<<

CLINICAL AND ANIMAL STUDIES OF -DERIVED PROTEIN MODIFICATIONS IN AUTISM, KIDNEY DIALYSIS, KERATITIS AND AGE-RELATED MACULAR DEGENERATION

by LIANG LU

Submitted in partial fulfillment of the requirements for the degree of Doctor of Philosophy

Thesis Advisor: Dr. Robert G. Salomon

Department of Chemistry CASE WESTERN RESERVE UNIVERSITY August 2007

CASE WESTERN RESERVE UNIVERSITY

SCHOOL OF GRADUATE STUDIES

We hereby approve the dissertation of

______

candidate for the Ph.D. degree *.

(signed)______(chair of the committee)

______

______

______

______

______

(date) ______

*We also certify that written approval has been obtained for any proprietary material contained therein.

This thesis is dedicated to my parents, my husband, my daughter, and my sisters.

iii TABLE OF CONTENTS Table of Contents iv List of Schemes ix List of Tables xi List of Figures xiv Acknowledgements xxiv List of Abbreviations and Acronyms xxvi Abstract xxxiii

CLINICAL AND ANIMAL STUDIES OF LIPID-DERIVED PROTEIN MODIFICATIONS IN AUTISM, KIDNEY DIALYSIS, KERATITIS AND AGE-RELATED MACULAR DEGENERATION

Chapter 1. Introduction 1

1.1. Oxidative stress and aging 2

1.2. Lipid oxidation 4

1.3. 4-Hydroxy-2-nonenal and its protein adducts 6

1.4. Levuglandins, isolevuglandins and their protein adducts 7

1.5. Oxidatively truncated phospholipids and carboxyalkylpyrrole modifications

of proteins 10

1.6. Carboxyethylpyrroles (CEPs) and their potential clinical applications 11

1.7. References 17

Chapter 2. Syntheses and Characterization of Carboxyethylpyrroles 27 2.1. Background 28

2.2. Results and Discussion 30

2.2.1. Paal-Knoor synthesis using 4,7-dioxoheptanoic acid is ineffective

iv for the preparation of CEPs 30

2.2.2. Synthesis of a 9-fluorenylmethyl (Fm) ester of DOHA 31

2.2.3. Syntheses of CEP-peptide and CEP-protein adducts by Paal-Knoor

synthesis with DOHA-Fm 31

2.2.4. CEP linked to proteins with an ω-aminohexanoyl tether 34

2.2.5. Syntheses of biotinylated CEP derivatives 36

2.2.6. Syntheses of ethanolamine phospholipid CEP derivatives 39

2.2.7. Synthesis of an active pentafluorophenyl ester of a lysyl CEP 39

2.2.8. Characterization of CEP-modified protein 40

2.2.9. Tertiary protein structures of CEP modified HSA 41

2.3. Conclusions 43

2.4. Experimental Procedures 44

2.5. References 70

Chapter 3. Lipid Oxidative Modifications in Autistic Disease 74

3.1. Background 75

3.2. Materials and Methods 82

3.3. Results and Discussion 84

3.3.1. CEP and iso[4]LGE2 protein adducts immunoreactivity in human plasma 84

3.3.2. Presence of anti-CEP and anti-iso[4]LGE2 autoantibodies in

human plasma 87

3.3.3. Protein bound nitrotyrosine, chlorotyrosine and bromotyrosine levels

in ASD and healthy controls 90

3.3.4. Correlations among lipid oxidative protein adducts and nitrative proteins 92

v 3.3.5. Correlations between autoantibodies and lipid oxidation

immunoreactivities 94

3.3.6. Lipid oxidation products and birth events in ASD patients 96

3.4. Conclusions 99

3.5. Experimental Procedures 101

3.6. References 112

Chapter 4. Serum E and Oxidative Protein Modification in

Hemodialysis 117

4.1. Background 118

4.2. Results 120

4.3. Discussion 128

4.4. Conclusions 133

4.5. Experimental Procedures 134

4.6. References 150

Chapter 5. Identification of Carboxyethylpyrrole

Phosphatidylethanolamine Adducts in vitro and in vivo 161

5.1. Background 162

5.2. Results and Discussion 168

5.2.1. Syntheses of authentic samples 168

5.2.2. Immunoreactivity of authentic CEP-PE 168

5.2.3. Identification of CEP-PE in oxidized lipid extracts 169

5.2.4. Identification of CEP-PE in lipid extracts that had not been

oxidized in vitro 170

vi 5.2.5. Quantification of CEP-PE in bovine retina 175

5.2.6. Identification of lysoCEP-PE in light-promoted oxidized lipid extracts 175

5.2.7. Identification of lysoCEP-PE in bovine retina 180

5.2.8. Identification of lysoCEP-PE in human plasma 180

5.2.9. Quantification of lysoCEP-PE in bovine retina and human plasma 183

5.3. Conclusions 185

5.4. Experimental Procedures 187

5.5. References 196

Chapter 6. Pilot Studies towards Identification of Levuglandin

Modified Proteins in Cornea 203

6.1. Introduction 204

6.1.1. LPS and inflammation 204

6.1.2. The cyclooxygenase and isoprostane pathways 204

6.1.3. LGE2 and iso[4]LGE2 bind avidly with proteins 207

6.1.4. Pyridoxamine, a potent trap that prevents protein modification by reactive

electrophiles 207

6.2. Results and Discussion 209

6.3. Experimental Procedures 212

6.4. References 214

Chapter 7. Syntheses of Isosteric Pyrazole Derivatives 221

7.1. Background 222

7.2. Results and Discussions 225

7.2.1. Synthesis of a 1,3-dione 225

vii 7.2.2. Synthesis and structure identification of 1-alkyl-5-pentylpyrazole-3-

carboxylic acid 225

7.2.3. Decarboxylation of a pyrazole carboxylic acid 227

7.2.4. Condensation of an alkylhydrazine with a β-ketoaldehyde 227

7.2.5. Attempted ozonolysis of an alkehylpyrazole 228

7.2.6. A strategy for conjugation of the isostere hapten with peptides 229

7.3. Experimental Procedures 230

7.4. References 236

Appendix 242

Bibliography 303

viii LIST of SCHEMES

Chapter 1

Scheme 1.1. Cyclooxygenase oxidation of AA generates PGs and LGs via rearrangement of PGH2. 8

Scheme 1.2. Free radical-induced oxidation of AA-PC produces isoLGs by rearrangement of isoP intermediates. 8

Scheme 1.3. Formation of LG-protein adducts, protein-protein and DNA-protein crosslinks. 10

Scheme 1.4. Oxidation of polyunsaturated fatty acids generates hydroxy-ω- oxoalkenoic acids, that react with proteins and form 2-(ω- carboxyalkyl)pyrroles. 12

Chapter 2

Scheme 2.1. DOHA exists in equilibrium with a spiroacylal hemiacetal. 30

Scheme 2.2. Synthesis of 9H-fluoren-9-ylmethyl ester 4,7-dioxo-heptanoic acid (DOHA-Fm, 2.4). 31

Scheme 2.3. Synthesis of CEP-dipeptide. 31

Scheme 2.4. Syntheses of CEP protein adducts. 33

Scheme 2.5. Syntheses of CEPs bound to proteins through a tether. 35

Scheme 2.6. Synthesis of biotinylated CEP derivative 2.21. 38

Scheme 2.7. Synthesis of biotinylated CEP derivative 2.25. 38

Scheme 2.8. Syntheses of ethanolamine phospholipid CEP derivatives. 39

Scheme 2.9. Synthesis of a lysyl CEP pentafluorophenyl derivative. 40

Chapter 5

Scheme 5.1. 4,5-(E)-Epoxy-2-(E)-heptenal reacts with phosphatidylethanolamine to produce pyrrole derivatives. 166

Scheme 5.2. Synthesis of CEP-PE and lysoCEP-PE. 168

ix Chapter 6

Scheme 6.1. Representative pathways showing and free radical induced generation of LGE2 and iso[4]LGE2 from AA-PC. 206

Scheme 6.2. Possible mechanism of pyrrole formation between pyridoxamine and 1,4-dicarbonyls. 208

Chapter 7

Scheme 7.1. Synthesis of 1-alkyl-5-pentylpyrazole through a decarboxylation route. 225

Scheme 7.2. General scheme of Barton decarboxylation. 227

Scheme 7.3. An alternative route for synthesis of pyrazole 7.5. 228

Scheme 7.4. Reductive amination of proteins with pyrazole aldehyde 7.6 that is to be generated by ozonolysis of alkene 7.5. 229

x LIST OF TABLES

Chapter 2

Table 2.1. Pyrrole concentration (μM) generated in 1 mg/mL CEPH-BSA prepared by coupling various initial protein concentrations with various CEPFmSu/Lys ratios. 36

Table 2.2. ELISA data for CEPH-BSA of Figure 2.1. 69

Table 2.3. ELISA data for CEP-HSA of Figure 2.1. 69

Chapter 3

Table 3.1. ELISA data for iso[4]LGE2-HSA standard for Figure 3.6. 103

Table 3.2. ELISA data for ASD patient (patient code A27) for Figure 3.6. 103

Table 3.3. ELISA data for control (normal code N11) for Figure 3.6. 104

Table 3.4. Data for CEP, iso[4]LGE2 immunoractivites and CEP, iso[4]LGE2 autoantibodies in ASD patients. 105

Table 3.5. Data for CEP, iso[4]LGE2 immunoractivites and CEP, iso[4]LGE2 autoantibodies in normal controls. 106

Table 3.6. Nitrotyrosine, chlorotyrosine and bromotyrosine levels in autistic patients. 109

Table 3.7. Nitrotyrosine, chlorotyrosine and bromotyrosine levels in normal controls. 110 Chapter 4

Table 4.1. Baseline clinical characteristics of patients. 120

Table 4.2. Plasma levels of alpha and gamma in placebo and treated patients. 122

Table 4.3. Plasma glycoxidation and protein-lipid oxidation products in placebo and vitamin E treated patients. 124

Table 4.4. Linear regression of glycoxidation and protein-lipid oxidation products with alpha or gamma tocopherol at baseline. 126

Table 4.5. Linear regression of iso[4]LGE2 with alpha or gamma tocopherol during placebo treatment at 3 months, 6 months and 9 months. 126

xi

Table 4.6. Linear regression of iso[4]LGE2 with alpha or gamma tocopherol during during treatment with α-tocopherol at 3 months, 6 months and 9 months. 127

Table 4.7. Linear regression matrix for glycoxidation and protein-lipid oxidation products at baseline. 127

Table 4.8. α-Tocopherol levels (μg/mL) in plasma from patients treated with vitamin E at baseline, three months, six months, nine months and twelve months. 136

Table 4.9. α-Tocopherol levels (μg/mL) in plasma from HD patients treated with placebo at baseline, three months, six months, nine months and twelve months. 137

Table 4.10. γ-Tocopherol levels (μg/mL) in plasma from patients treated with vitamin E at baseline, three months, six months, nine months and twelve months. 138

Table 4.11. γ-Tocopherol levels (μg/mL) in plasma from HD patients treated with placebo at baseline, three months, six months, nine months and twelve months. 138

Table 4.12. Pentosidine levels (pmol/mg protein) in plasma from HD patients treated with vitamin E supplementation at baseline, three months, six months, nine months and twelve months. 140

Table 4.13. Pentosidine levels (pmol/mg protein) in plasma from HD patients treated with placebo at baseline, three months, six months, nine months and twelve months. 141

Table 4.14. HNE-derived pentylpyrrole immunoreactivities in plasma from patients treated with vitamin E at baseline, 3 months, 6 months, 9 months and 12 months. 144

Table 4.15. HNE-derived pentylpyrrole immunoreactivities (nmol/mL) in plasma from patients treated with placebo at baseline, 3 months, 6 months, 9 months and 12 months. 145

Table 4.16. Iso[4]LGE2-protein immunoreactivities (nmol/mL) in plasma from patients treated with vitamin E at baseline, 3 months, 6 months, 9 months and 12 months. 146

xii Table 4.17. Iso[4]LGE2-protein immunoreactivities (nmol/mL) in plasma from patients treated with placebo at baseline, 3 months, 6 months, 9 months and 12 months. 147

Table 4.18. ONE-protein immunoreactivities (pmol/mg protein) in plasma from patients treated with vitamin E at baseline, 3 months, 6 months and 9 months. 148

Table 4.19. ONE-protein immunoreactivities (pmol/mg protein) in plasma from patients treated with placebo at baseline, 3 months, 6 months and 9 months. 149

Chapter 5

Table 5.1. Distribution of the fatty acids of PE and PC in human ROS (as mol% of total fatty acid). 162

Table 5.2. ELISA data for CEP-PE in Figure 5.1. 188

Table 5.3. ELISA data for CEP-CEO in Figure 5.1. 188

Chapter 7

Table 7.1. HMQC and HMBC data for 7.4 (in parts per million). 226

Appendix

Table S1. Total iron levels of patients treated with vitamin Eα or placebo at baseline, three months, six months and nine months. 292

Table S2. Erythropoietin administrated in patients treated with vitamin Eα or placebo at baseline, three months, six months and nine months. 293

Table S3. Ferritin levels of patients treated with vitamin Eα or placebo at baseline, three months, six months and nine months. 294

xiii LIST OF FIGURES

Chapter 1

Figure 1.1. The sources and cellular responses to reactive oxygen species (ROS). 3

Figure 1.2. CEP-HSA as well as dipeptide (Ac-gly-lys-OH) induces angiogenesis in a rat corneal micropocket assay. (A) HSA (1 μg), (B) CEP-HSA (1 μg), (C) dipeptide (41 ng), (D) CEP-dipeptide (37 ng). 13

Figure 1.3. Outer retinal pathology present in mice immunized with CEP-MSA (A-J). Large arrows indicate several inflammatory cells immediately adjacent to the RPE or in the IPM. Large empty vacuoles are also evident (C-I). Some of which appear to be intracellular (I). In (J) the RPE has degenerated (asterisks). Bar in lower right of (J) represent 25 μm. 14

Chapter 2

Figure 2.1. Inhibition curves showing cross-reactivity of the anti-CEP-KLH antibody for CEP-HSA (▲) and CEPH-BSA (●) against CEP-HSA as coating agent. 36

Figure 2.2. A ball-and-stick model of 3D structure for human serum albumin. All the lysines are shown as space-filling structures in the left unit. Only lysines that become CEP modified are shown in the right unit. The lysine in the sequence which is common to HSA, BSA and MSA is marked. 42

Chapter 3

Figure 3.1. Anti-CEP staining of brain cortical regions from 3 of the control cases aged 5, 6 and 11 years (A, B, C) and 3 autism cases aged 9, 7, and 5 years (D, E, F). 80

Figure 3.2. CEP immunoreactivity detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 84

Figure 3.3. Levels of individual CEPs in plasma from 27 patients with diagnosed ASD and 11 healthy controls. 85

Figure 3.4. Iso[4]LGE2 immunoreactivity detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels

xiv detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 85

Figure 3.5. Levels of individual iso[4]LGE2-protein adducts in plasma from 27 patients with diagnosed ASD and 11 healthy controls. 86

Figure 3.6. Inhibition curves for binding of anti-iso[4]LGE2-KLH to iso[4]LGE2- BSA by iso[4]LGE2-HSA (●), plasma from an ASD patient (■), and a normal control (▲). 86

Figure 3.7. CEP autoantibody titer detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 88

Figure 3.8. Levels of individual individual CEP autoantibody titer in plasma from 26 patients with diagnosed ASD and 5 healthy controls. 88

Figure 3.9. Iso[4]LGE2 autoantibody titer detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 89

Figure 3.10. Levels of individual iso[4]LGE2 autoantibody titer in plasma from 27 patients with diagnosed ASD and 11 healthy controls. 89

Figure 3.11. Nitrotyr/Tyr ratio detected in human plasma from (◆) ASD and (▲) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 91

Figure 3.12. Cltyr/Tyr ratio detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 91

Figure 3.13. Brtyr/Tyr ratio detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set. 92

Figure 3.14. Correlation between CEP immunoreactivity and iso[4]LGE2 immunoreactivity in human plasma from (●) ASD and (∆) normal controls. 93

Figure 3.15. Correlation between iso[4]LGE2 and CEP immunoreactivities in human plasma from (◆) ASD and (■) normal controls. The horizontal and vertical dashed lines indicate the mean values of CEP

xv and iso[4]LGE2 immunoreactivities, respectively. Out of 27 ASD patients, 6 (22%) are in region IV while 1 out of 11 normal controls (9%) is in region IV. ASD patients account for 86% (6/7) of the data in region IV. 93

Figure 3.16. Correlation between CEP immunoreactivity and nitrotyro/Tyr ratio in human plasma from (●) ASD and (∆) normal controls. 94

Figure 3.17. Correlation between iso[4]LGE2 immunoreactivity and nitrotyro/Tyr ratio in human plasma from (●) ASD and (∆) normal controls. 94

Figure 3.18. Correlation between CEP autoantibody titer and iso[4]LGE2 autoantibody titer in human plasma from (●) ASD and (∆) normal controls. 95

Figure 3.19. Correlation between CEP immunoreactivity and CEP autoantibody titer in human plasma from (●) ASD and (∆) normal controls. 95

Figure 3.20. Correlation between iso[4]LGE2 immunoreactivity and iso[4]LGE2 autoantibody titer in human plasma from (●) ASD and (∆) normal controls. 96

Figure 3.21. CEP immunoreactivity grouped according to birth events of ASD patients with no events, born premature or other events birth events. 97

Figure 3.22. CEP autoantibody titer grouped according to birth events of ASD patients with no events, born premature or other events birth events. 97

Figure 3.23. Iso[4]LGE2 immunoreactivity grouped according to birth events of ASD patients with no events, born premature or other events birth events. 98

Figure 3.24. Iso[4]LGE2 autoantibody titer grouped according to birth events of ASD patients with no events, born premature or other events birth events. 98

Chapter 4

Figure 4.1. Relationship between plasma levels of Vitamin E alpha and gamma before and after treatment. A. Baseline R = 0.46, p = 0.0055. B. The positive correlation between vitamin E alpha and gamma is present in the placebo group, left regression line (R = 0.42, p = 0.05). In the patients treated with vitamin E, there is a negative correlation between the two metabolites, right regression line (R = 0.11, “not significant”). Gray symbols, placebo. Black symbols, Vitamin E treatment. Open circles represent baseline; squares, 3 month values. 123

xvi

Figure 4.2. Levels of HNE-derived pentylpyrrole over the course of study. 125

Figure 4.3. Levels of iso[4]LGE2-protein adduct over the course of study. 125

Figure 4.4. Fenton and Fenton-like reactions. 131

Figure 4.5. Reduction of transition metal ions by . 131

Chapter 5

Figure 5.1. Inhibition curves showing crossreactivity of the anti-CEP-KLH antibody for CEP-CEO (y) and CEP-PE (ƒ) against CEP-CEO as coating agent. 169

Figure 5.2. Proposed MRM fragments of CEP-PE and corresponding m/z. 171

Figure 5.3. Daughter scan of CEP-PE. 171

Figure 5.4. MRM chromatograms of CEP-PE. 172

Figure 5.5. MRM of lipid extracts from bovine retina after UV light promoted oxidation. A single peak with a retention time that is identical to that of authentic standard CEP-PE was present in the MRM chromatogram of oxidative lipid at each parent/daughter ion pair channel. 173

Figure 5.6. MRM of bovine retinal extracts that had not been oxidized in vitro. A single peak with a retention time that is identical to that of authentic CEP-PE is present in the MRM chromatogram monitoring the 838.3 → 255.4 and 838.3 → 391.2 transitions. 174

Figure 5.7. Standard curve and calibration equation for CEP-PE. 175

Figure 5.8. Proposed fragments of lysoCEP-PE and corresponding m/z. 177

Figure 5.9. Daughter scan of lysoCEP-PE. 177

Figure 5.10. MRM chromatograms of lysoCEP-PE. Pure lysoCEP-PE (10 ng/μL in methanol) was injected onto a Shimadzu LC-10AD HPLC system, eluting with 1 mM ammonium acetate in 20% acetonitrile and using a linear gradient to 100% acetonitrile over 3 minutes, followed by 100% acetonitrile for 10 minutes. 178

xvii Figure 5.11. MRM of bovine retinal after UV-promoted oxidation. Lipid extracts (50 ng/μL in methanol) was injected onto a Shimadzu LC- 10AD HPLC system as described in Figure 5.10. A single peak with a retention time that is identical to that of authentic lysoCEP-PE was present in the MRM chromatogram of oxidized lipid in each parent/daughter ion pair channel. 179

Figure 5.12. MRM chromatograms of authentic lysoCEP-PE. Pure lysoCEP-PE (50 ng) was injected onto a Waters 2790 HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes. Note: This is a different system than Figures 5.10-5.11 and retention times are, therefore, different. 181

Figure 5.13. MRM chromatograms of lipid extracts from a plasma sample of a normal control. Lipid extracts (150 μg in 20 μL methanol) was injected onto a Waters 2790 HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes. 182

Figure 5.14. MRM chromatograms of lipid extracts from a plasma sample of an AMD patient. Lipid extracts (100 μg in 20 μL methanol) was injected onto a Waters 2790 HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes. 183

Figure 5.15. Standard curve and calibration equation of authentic lysoCEP-PE. 184

Chapter 6

Figure 6.1. Eicosanoid synthesis. 206

Figure 6.2. Cornea of C57BL/6 mouse was treated in vivo with LPS or PBS for 24 h by injection or scratch. Sections (5 μm) of cornea were stained with LGE2 and iso[4]LGE2 pAb. A, B, C, J, K, L – Untreated; D, E, F – PBS injection; G, H, I – LPS injection; M, N, O – PBS treated (scratch); P, Q, R – LPS treated (scratch). Panels treated with preimmune, LGE2 and iso[4]LGE2 pAbs are indicated in the Figure. 210

Chapter 7

Figure 7.1. Pyrroles cross-link with proteins. 224

Figure 7.2. Structural description of C – H HMBC coupling for both 3-carboxyl- 5-pentyl pyrazole and 3-pentyl-5-carboxyl pyrazole. 226

xviii

Appendix

1 Figure S1. H NMR (200 MHz, CDCl3) spectrum of methyl 6-(2,5-dioxolanyl) -4-oxohexanoate (2.1). 243

13 Figure S2. C NMR (50 MHz, CDCl3) spectrum of methyl 6-(2,5-dioxolanyl) -4-oxohexanoate (2.1). 243

1 Figure S3. H NMR (200 MHz, CDCl3) spectrum of 6-(2,5-dioxolanyl)-4- oxohexanoic acid (2.2). 244

13 Figure S4. C NMR (100 MHz, CDCl3) spectrum of 6-(2,5-dioxolanyl)-4- oxohexanoic acid (2.2). 244

1 Figure S5. H NMR (200 MHz, CDCl3) spectrum of (2.3). 245

13 Figure S6. C NMR (50 MHz, CDCl3) spectrum of (2.3). C, CH2 (-); CH, CH3 (+). 245

1 Figure S7. H NMR (200 MHz, CDCl3) spectrum of DOHAFm (2.4). 246

13 Figure S8. C NMR (100 MHz, CDCl3) spectrum of DOHAFm (2.4). 246

1 Figure S9. H NMR (400 MHz, CDCl3) spectrum of (2.5). 247

13 Figure S10. C NMR (100 MHz, CDCl3) spectrum of (2.5). 247

1 Figure S11. H NMR (400 MHz, CDCl3) spectrum of (2.6). 248

13 Figure S12. C NMR (150 MHz, CDCl3) spectrum of (2.6). 248

1 Figure S13. H NMR (400 MHz, CDCl3) spectrum of (2.9). 249

13 Figure S14. C NMR (100 MHz, CDCl3) spectrum of (2.9). 249

1 Figure S15. H NMR (400 MHz, CDCl3) spectrum of CEPFmSu (2.10). 250

13 Figure S16. C NMR (100 MHz, CDCl3) spectrum of CEPFmSu (2.10). 250

1 Figure S17. H NMR (400 MHz, CD3OD) spectrum of (2.18). 251

1 Figure S18. H NMR (200 MHz, CD3OD) spectrum of (2.19). 251

1 Figure S19. H NMR (200 MHz, CDCl3) spectrum of (2.20). 252

xix 1 Figure S20. H NMR (400 MHz, CD3OD) spectrum of (2.21). 252

1 Figure S21. H NMR (200 MHz, CDCl3) spectrum of (2.22). 253

1 Figure S22. H NMR (400 MHz, CD3OD) spectrum of (2.23). 253

13 Figure S23. C NMR (100 MHz, CD3OD) spectrum of (2.23). 254

1 Figure S24. H NMR (400 MHz, CD3OD) spectrum of (2.24). 254

1 Figure S25. H NMR (400 MHz, CD3OD) spectrum of (2.25). 255

13 Figure S26. C NMR (100 MHz, CD3OD) spectrum of (2.25). 255

1 Figure S27. H NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.26). 256

13 Figure S28. C NMR (50 MHz, CDCl3:CD3OD = 1:1) spectrum of (2.26). 256

1 Figure S29. H NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.27). 257

13 Figure S30. C NMR (100 MHz, CDCl3:CD3OD = 1:1) spectrum of (2.27). 257

1 Figure S31. H NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.28). 258

13 Figure S32. C NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.28). 258

1 Figure S33. H NMR (400 MHz, CDCl3) spectrum of (2.29). 259

13 Figure S34. C NMR (100 MHz, CDCl3 :CD3OD:D2O = 50:50:1) spectrum of (2.29). 259

1 Figure S35. H NMR (400 MHz, CDCl3) spectrum of (2.30). 260

13 Figure S36. C NMR (100 MHz, CDCl3) spectrum of (2.30). 260

1 Figure S37. H NMR (400 MHz, CDCl3) spectrum of (2.31). 261

13 Figure S38. C NMR (100 MHz, CDCl3) spectrum of (2.31). 261

Figure S39. Tandem MS characterization of CEP modified HSA shows series of fragment ions sufficient to identify CEP modifications on lysyl residues of the HSA peptides. (A). MS/MS spectrum of the doubly charged ion m/z 589.266. (B). MS/MS spectrum of the doubly charged ion m/z 674.762. (C). MS/MS spectrum of the doubly charged ion m/z 881.401. (D). MS/MS spectrum of the doubly charged ion m/z 687.784. (E). MS/MS spectrum of the doubly

xx charged ion m/z 636.252. Asterisks denote fragment ions with the modified lysyl residue. 263

Figure S40. Tandem MS characterization of CEP modified MSA. (A). MS/MS spectrum of the doubly charged ion m/z 650.3296. (B). MS/MS spectrum of the doubly charged ion m/z 1047.0802. (C). MS/MS spectrum of the doubly charged ion m/z 769.4069. (D). MS/MS spectrum of the doubly charged ion m/z 612.8407. (E). MS/MS spectrum of the doubly charged ion m/z 872.9611. (F). MS/MS spectrum of the triplet charged ion m/z 849.4302. (G). MS/MS spectrum of the triplet charged ion m/z 857.4633. (H). MS/MS spectrum of the triplet charged ion m/z 694.6888. Asterisks denote fragment ions with the modified lysyl residue. 266

Figure S41. Tandem MS characterization of CEP modified CEO. (A). MS/MS spectrum of the doubly charged ion m/z 540.2886. (B). MS/MS spectrum of the triplet charged ion m/z 801.7021. (C). MS/MS spectrum of the doubly charged ion m/z 839.3332. (D). MS/MS spectrum of the triplet charged ion m/z 808.0259. (E). MS/MS spectrum of the doubly charged ion m/z 610.2589. (F). MS/MS spectrum of the doubly charged ion m/z 630.8334. (G). MS/MS spectrum of the doubly charged ion m/z 459.1887. Asterisks denote fragment ions with the modified lysyl residue. 269

Figure S42. Tandem MS characterization of CEP modified myoglobin. (A). MS/MS spectrum of the doubly charged ion m/z 671.2667. (B). MS/MS spectrum of the doubly charged ion m/z 604.9635. (C). MS/MS spectrum of the doubly charged ion m/z 815.2087. (D). MS/MS spectrum of the quartet charged ion m/z 557.4288. (E). MS/MS spectrum of the doubly charged ion m/z 785.6061. (F). MS/MS spectrum of the doubly charged ion m/z 899.6666. Asterisks denote fragment ions with the modified lysyl residue. 272

Figure S43. Tandem MS characterization of CEP modified GPDH. (A). MS/MS spectrum of the doubly charged ion m/z 655.3343. (B). MS/MS spectrum of the doubly charged ion m/z 669.3289. (C). MS/MS spectrum of the doubly charged ion m/z 923.9847. (D). MS/MS spectrum of the doubly charged ion m/z 639.3413. (E). MS/MS spectrum of the doubly charged ion m/z 740.8362. Asterisks denote fragment ions with the modified lysyl residue. 274

Figure S44. Tandem MS characterization of CEPH modified BSA. (A). MS/MS spectrum of the triplet charged ion m/z 594.9934. (B). MS/MS spectrum of the doubly charged ion m/z 541.8060. (C). MS/MS spectrum of the doubly charged ion m/z 937.9577. (D). MS/MS spectrum of the doubly charged ion m/z 526.7835. (E). MS/MS

xxi spectrum of the doubly charged ion m/z 612.3604. (F). MS/MS spectrum of the doubly charged ion m/z 689.4319. (G). MS/MS spectrum of the doubly charged ion m/z 765.3984. (H). MS/MS spectrum of the doubly charged ion m/z 964.5088. Asterisks denote fragment ions with the modified lysyl residue. 278

Figure S45. Tandem MS characterization of CEPH modified BSA. (A). MS/MS spectrum of the doubly charged ion m/z 512.2829. (B). MS/MS spectrum of the doubly charged ion m/z 627.8604. (C). MS/MS spectrum of the doubly charged ion m/z 608.8177. (D). MS/MS spectrum of the doubly charged ion m/z 732.3707. Asterisks denote fragment ions with the modified lysyl residue. 280

Figure S46. Tandem MS characterization of the doubly charged ion m/z 596.8715 from a MS scan of tryptic digested CEPH modified CEO. Asterisks denote fragment ions with the modified lysyl residue. 281

Figure S47. Tandem MS characterization of CEPH modified GPDH. (A). MS/MS spectrum of the doubly charged ion m/z 635.0367. (B). MS/MS spectrum of the doubly charged ion m/z 712.5500. (C). MS/MS spectrum of the doubly charged ion m/z 919.7650. (D). MS/MS spectrum of the doubly charged ion m/z 572.9763. (E). MS/MS spectrum of the triplet charged ion m/z 899.6923. (F). MS/MS spectrum of the doubly charged ion m/z 726.0422. (G). MS/MS spectrum of the doubly charged ion m/z 980.7797. (H). MS/MS spectrum of the doubly charged ion m/z 731.1005. (I). MS/MS spectrum of the doubly charged ion m/z 941.7422. (J). MS/MS spectrum of the triplet charged ion m/z 579.9560. (K). MS/MS spectrum of the doubly charged ion m/z 508.9000. (L). MS/MS spectrum of the doubly charged ion m/z 696.1018. (M). MS/MS spectrum of the doubly charged ion m/z 797.5823. Asterisks denote fragment ions with the modified lysyl residue. 286

Figure S48. Tandem MS characterization of CEPFmHSA revealed five CEP modifications. (A). MS/MS spectrum of the doubly charged ion m/z 636.248. (B). MS/MS spectrum of the doubly charged ion m/z 881.408. (C). MS/MS spectrum of the doubly charged ion m/z 571.262. (D). MS/MS spectrum of the doubly charged ion m/z 687.787. (E). MS/MS spectrum of the doubly charged ion m/z 674.765. 289

Figure S49. Tandem MS characterization of CEPFmHSA revealed five CEPFm modifications. (A). MS/MS spectrum of the doubly charged ion m/z 725.281. (B). MS/MS spectrum of the doubly charged ion m/z 660.289. (C). MS/MS spectrum of the doubly charged ion m/z 776.820. 291

xxii

1 Figure S50. H NMR (300 MHz, CDCl3) spectrum of (7.1). 295

13 Figure S51. C NMR (75 MHz, CDCl3) spectrum of (7.1). 295

1 Figure S52. H NMR (300 MHz, CDCl3) spectrum of (7.2). 296

13 Figure S53. C NMR (75 MHz, CDCl3) spectrum of (7.2). 296

1 Figure S54. H NMR (300 MHz, CDCl3) spectrum of (7.3). 297

1 Figure S55. H NMR (300 MHz, CDCl3) spectrum of (7.4). 297

13 Figure S56. C NMR (75 MHz, CDCl3) spectrum of (7.4). 298

1 Figure S57. H NMR (400 MHz, CDCl3) spectrum of (7.5). 298

13 Figure S58. C NMR (100 MHz, CDCl3) spectrum of (7.5). 299

Figure S59. HMQC of 3-carboxyl-5-pentyl pyrazole (7.4). 299

Figure S60. HMBC of 3-carboxyl-5-pentyl pyrazole (7.4). 300

1 Figure S61. H NMR (300 MHz, CDCl3) spectrum of (7.7). 300

1 Figure S62. H NMR (300 MHz, CDCl3) spectrum of (7.8). 301

1 Figure S63. H NMR (300 MHz, CDCl3) spectrum of (7.9). 301

1 Figure S64. H NMR (400 MHz, CDCl3) spectrum of (7.10). 302

xxiii Acknowledgements

First of all, I would like to express my deepest sense of gratitude and everlasting

respect to my research advisor Dr. Robert G. Salomon for his invaluable guidance,

fruitful discussions, strictly scientific attitude, and humane understanding throughout my

graduate studies. He provided me with the best education that an advisor can bring to his

students. He encouraged me to never accept anything short of my personal best in my

research. I am also deeply moved by his noble personality. His intelligence, his kindness,

and his generosity make me very thankful to be one of his students. I wish I could stay

longer to experience more of his guidance, both academically and personally.

I am very grateful to my committee members: Dr. Pearson, Dr. Zagorski, and Dr.

Burda for their precious time, efforts, suggestions, and support in my candidate

qualification and thesis. Special thanks to Dr. Weiss for being on my committee, and for

her efforts, intelligence and time on our paper and my thesis.

It is my pleasure to acknowledge Dr. John W. Crabb, Dr. Joe Hollyfield, Dr.

Victor Perez, and their groups, at the Cole Eye Institute, Cleveland Clinic Foundation, for

their collaboration. Special thanks are due to Dr. Xiaorong Gu for her valuable help and

sisterly care.

I would like to thank Dr. Lian Shan, at Frantz Biomarkers, for all the help and instruction on mass spectrometry.

I would like to thank Dr. Eric Pearlman and his group at the Center for Global

Health & Diseases, for their training on immunostaining techniques.

I would like to thank Dr. Salomon’s group, former and current members: Dr.

Bogdan Gugiu, Dr. Suresh Annangudi, Dr. James Laird, and Ken, Wujuan, Jawoo, Xi, xxiv Wei, Xiaodong, Yunfeng, Li, and Hua for many thoughtful discussions and the overall friendly atmosphere. Special thanks to Jim, for all his assistance in my effort to improve my writing and pronunciation.

I would also like to thank Dr. Dale Ray and Dr. Jim Faulk and other staff members in Department of Chemistry in Case Western Reserve University for their help on numerous occasions.

I greatly appreciate Jun Li, Ming Zhang, Liqing Ma and all my friends for all the beautiful memories we had together. Their friendship made my stay at U.S.A. pleasant, enjoyable and deeply cherished.

Finally, I would like to express my deepest gratitude to my parents, my husband, my sisters and their family, and my sweety heart--my daughter-- for their endless love, support, and countless sacrifices.

Liang Lu

xxv LIST OF ABBREVIATIONS AND ACRONYMS

Abbreviations and Acronyms Equivalent

AA Arachidonyl acid

AA-PC Arachidonic phosphatidylcholine

AGE Advanced glycation end product

ALPE Advanced lipid peroxidation end product

AMD Age-related macular degeneration

AOPP Advanced oxidative protein product

APT Attached proton test

AS Atherosclerosis

ASD Autism spectrum disorders

BCA Bicinchoninic acid

BHT

BrTyr Bromotyrosine

BSA Bovine serum albumin

CAT Catalase

CDCl3 Deutrated

CD3OD Deutrated methanol

γ-CEHC 2,7,8-Trimethyl-2-(β-carboxyethyl)-6-

hydroxychroman

CEO Chicken egg ovulbumin

CEP 2-(ω-Carboxyethyl)pyrrole

xxvi CEPH Carboxyethylpyrrole derivatives of 6-

aminohexanoic acid

CEP-PE Carboxyethylpyrrole

phosphatidylethanolamine adducts

CHAOS Cambridge Heart Study

CHP 2-(ω-Carboxyheptyl)pyrrole

ClTyr Chlorotyrosine

CME Carboxymethyl-PE

CPP 2-(ω-Carboxypropyl)pyrrole

COX Cyclooxygenase

DBF Dibenzofulvene

DBU 1,8-Diazabicyclo[5.4.0] undec-7-ene

DCC Dicyclohexylcarbodiimide

DHA Docosahexaenoic acid

DAPI 4,6-Diamidino- -2-phenylindole

DMAP 4-N,N-Dimethylaminopyridine

DMF Dimethyl formide

DNA Desoxyribonucleic acid

D2O Deutrated water

DOHA 4,7-Dioxoheptanoic acid

DOHA-Fm 9H-fluoren-9-ylmethyl ester of DOHA

DPCs DNA-protein cross-links

xxvii DSM Diagnostic and Statistical Manual of Mental

Disorders

EDTA Ethylenediaminetetraacetic acid

EI Electron impact

ELISA Enzyme-linked immunosorbent assay

ESRD End stage renal disease

FAB Fast atom bombardment

FCS Fetal calf serum

Fm 9-Fluorenylmethyl

GISSI Gruppo Italiano per lo Studio della

Sopravvivenza nell’Infarto miocardio

GPDH Glycerol-3-phosphate dehydrogenase

GPx peroxidase

GSH Reduced glutathione

GSSH Glutathione-glutathione

GTP Guanosine triphosphate pHA p-Hydroxyphenylacetaldehyde

HD Hemodialysis

HDDE 4-Hydroxydodeca-(2E,6Z)-dienal

HEPE N-(Hexanoyl)phosphatidylethanolamine HHE 4-Hydroxy-2E- hexenal HMBC Heteronuclear multiple bond coherence

HMQC Heteronuclear multiple quantum coherence

xxviii HNE 4-Hydroxy-2-nonenal

HODA 9-Hydroxy-12-oxo-10(E)-dodecenoic acid

HOHA 4-Hydroxy-7-oxohept-5-enoic acid

HODA-PC 1-Palmitoyl-2-(9-hydroxy-12-oxododec-10- enoate)-sn-glycero-3-phosphatidylcholine

HOOA 5-Hydrox-8-oxo-6-octenoic acid

HOPE Heart Outcomes Prevention Evaluation

HPLC High performance liquid chromatography

HRMS High resolution mass spectrometry

HSA Human serum albumin

IFN-γ Interferon-γ

IL-2 Interleukin-2

IPM Interphotoreceptor matrix iso[4]LGE2 Iso[4]levuglandin E2 isoLGE2 Isolevuglandin E2 isoP Isoprostane

KLH Keyhole limpet hemocyanin

LA Linoleic acid

LC Liquid chromatography

LGD2 Levuglandin D2

LGE2 Levuglandin E2

LDL Low density lipoprotein

LF Lipofuscin

xxix LG Levuglandin

LOOH Lipid hydroperoxide

LPS Lipopolysaccharide

Lyso-PC 1-Palmityl-2-hydroxy-sn-glycero-3- phosphatidylcholine

Lyso-PE 1-Palmityl-2-hydroxy-sn-glycero-3-phospho- - ethanolamine MALDI-TOF Matrix assisted laser desorption ionization- time of flight m-CPBA m-Chloroperbenzoic acid

MCP-1 Monocyte chemoattractant protein-1

MDA Malondialdehyde

MHC Major histocompatibility complex

MHz Megahertz

MMR Measles-mumps-rubella

M-PE 1-Myristyl-2-hydroxy-sn-glycero-3-

phosphatidylethanolamine

MPM Mouse peritoneal macrophage

MPO Myeloperoxidase

MRM Multiple reaction monitoring

MS Mass spectrometry

MSA Mouse serum albumin

MS/MS Tandem mass spectrometry

MT Metallothionein

NMR Nuclear magnetic resonance

xxx NO

NOS Nitric oxide synthase

ONE 4-Oxo-2-nonenal

ON 9-Oxononanyl

OV 2-Oxovaleryl oxLDL Oxidized low density lipoprotein oxPC Oxidized phosphatidylcholines oxPL Oxidized phospholipids

PAGE Polyacrylamide gel electrophoresis

PBS Phosphate buffered saline

PC Phosphatidylcholine

PDD Pervasive developmental disorders

PE Phosphatidylethanolamine

PG Prostaglandin

PGD2 Prostaglandin endoperoxide D2

PGE2 Prostaglandin endoperoxide E2

PGH2 Prostaglandin endoperoxide H2

PL Phospholipids

POPE 1-Palmityl-2-oleyl-sn-glycero-3- phosphoethanolamine PP 2-Pentylpyrrole ppm Parts per million

PTCA Percutaneous transluminal coronary

angioplasty

xxxi PUFA Polyunsaturated fatty acid

QTOF Quadrupole time-of-flight

Rf Retention factor

ROS Rod outer segment

ROxS Reactive oxygen species

RPE Retina pigment epithilium

S.D. Standard deviation

SDS/PAGE Sodium dodecyl sulfate/ polyacrylamide gel electrophoresis

SH Sulfhydryl

SO Superoxide

SOD Superoxide dismutase

Su N-Hydroxysuccinimide

TCA Trichloroacetic acid

TLC Thin layer chromatography

TNF Tumor necrosis factor

Tyr Tyrosine

α-TTP α-Tocopherol transfer protein

UV Ultraviolet

xxxii Clinical and Animal Studies of Lipid-Derived Protein Modifications in Autism, Kidney Dialysis, Keratitis and Age-Related Macular Degeneration

Abstract

By

LIANG LU

Lipid oxidation in diverse biological systems contributes to normal physiological

processes and is increasingly believed to be associated with age-related diseases. Lipid

oxidation products, e.g., isolevuglandins and γ-hydroxyalkenals, and their protein adducts

are produced during and may even contribute to disease pathogenesis. This thesis reports

clinical studies on the occurrence of 2-pentylpyrrole, iso[4]LGE2-protein adducts and

carboxyethylpyrrole (CEP) protein modifications in brain and blood from autistic

individuals, blood from hemodialysis patients, as well as cornea from a mouse model of

keratitis. Similar levels of these modifications and autoantibodies against them were

detected in blood from both autistic individuals and normal controls. Autistic patients born prematurely showed significantly elevated iso[4]LGE2-protein adduct levels. Oral

supplementation with α-tocopherol had no significant effect on levels of oxidative

protein modifications in patients treated by hemodialysis, and thus, does not result in an

antioxidant benefit in hemodialysis. We detected the formation of iso[4]LGE2- and

LGE2-protein adducts upon injection of lipopolysaccharide into mouse cornea, a model of

keratitis.

xxxiii We anticipated that docosahexaenoic acid-derived oxidatively truncated

phospholipids containing reactive electrophilic 4-hydroxy-7-oxohept-5-enoates (HOHA)

would convert the primary amino group of PEs into carboxyethylpyrrole phosphatidylethanolamine adducts (CEP-PEs). I detected a low level of CEP-PE in a bovine retinal lipid extract and quantified the generation of CEP-PE adduct and a CEP- modified lysophospholipid, lysoCEP-PE, upon UV light-promoted oxidation of the retinal lipid extract. Similar levels, about 1.2 ng/mL, of lysoCEP-PE were detected in plasma samples from a normal control and a patient with age-related macular degeneration (AMD).

A reliable source of CEP-modified proteins was needed to provide: (1) reagents for immunoassays that measure levels of CEPs or anti-CEP autoantibodies as biomarkers

for clinical prognosis of AMD, and (2) antigens to test the hypothesis that immune responses against CEP-protein adducts generated in the retina contribute to the pathogenesis of AMD, (3) for studies on the stimulation of angiogenesis by CEPs and its inhibition, and (4) for the production of CEP-PEs as standards for analyses of these potential markers of oxidative injury. An efficient, reliable synthesis of CEPs was

developed that exploits a flourenylmethyl ester derivative of HOHA as a key reagent.

xxxiv

Chapter 1

Introduction

1 1.1 Oxidative stress and ageing.

Living in an oxygenated environment has required the evolution of effective cellular

strategies to detect and detoxify metabolites of molecular oxygen known as reactive

oxygen species. The appropriate and inappropriate production of oxidants, together with the ability of organisms to respond to oxidative stress, is intricately connected to ageing and life span.

------Toren Finkel and Nikki J. Holbrook

Aging has been characterized as “the progressive accumulation of changes with time

that are associated with or responsible for the ever-increasing susceptibility to disease and

death which accompanies advancing age.”1 In 1956, Denham Harman proposed his

famous free radical theory of aging which speculated that free oxygen radicals produced

during aerobic respiration cause cumulative oxidative damage, resulting in aging and

death.2 Results from disparate experimental systems have recently shown that reactive

oxygen species (ROxS) production provides the strongest correlation with overall

longevity: the greater the production, the shorter the life span.3-6 Whether or not they

determine life span, it is becoming apparent that oxygen radicals are certainly important

players in aging’s pathophysiology.

ROxS include a variety of diverse chemical species such as superoxide anions,

hydroxyl radicals and hydrogen . These radical species can either be generated

2 exogenously or produced intracellularly from several different sources (Figure 1.1).

Figure 1.1. The sources and cellular responses to reactive oxygen species (ROxS).7

Although free radicals may certainly cause harm to biological processes, there is increasing evidence that products of free radical-induced lipid oxidation serve as specific signaling molecules under both physiological and pathophysiological conditions.8-11 For example, ROxS produced in response to stimulation by growth factors participate in regulating the proliferative response.12 The immune system utilizes some of these

3 products to mark foreign invaders or damaged tissue, and thus indicate which tissues

need to be removed from the body.13, 14 As a result, an elevation in intracellular oxidant

levels has two potentially important effects: damage to various cell components and triggering of the activation of specific signaling pathways.7 Both of these effects are

related to numerous cellular processes linked to aging and the development of age-related diseases.

An intricate enzymatic and non-enzymatic antioxidant defence system encompassing catalase (CAT), superoxide dismutase (SOD) and (GPx), as well as small antioxidant molecules such as tocopherols, flavonoids, ascorbic acids, and , restrains and regulates overall ROxS levels to maintain physiological homeostasis.15, 16 Perturbation of the physiological role of oxidants in cellular

proliferation and host defence might be caused by lower ROxS levels than the

homeostatic set point. Meanwhile, high ROxS levels may result in cell death or accelerate

aging and age-related diseases.7 This is traditionally referred to as oxidative stress.

Consequences of this stress include modification to cellular proteins, lipids and DNA.

1.2. Lipid oxidation.

Oxygen-dependent deterioration of lipids has been observed since antiquity as a big

problem in the storage of oils (mainly olive oil). The first studies of this lipid oxidation

problem were those of the Swiss chemist Nicolas-Theodore de Saussure who noticed around 1800 that oil became viscous and had a bad smell when exposed to air.17

4 Systematic studies of lipid autoxidation may be regarded to have initiated around the

1940s since Criegee et al.18 proposed that hydroperoxides are the primary products of

hydrocarbon oxidation. In 1961, Bolland19 documented that the primary autoxidation

products of linoleic acid are hydroperoxides (on carbon atom 9 or 13) containing

conjugated dienes. The free radical-induced oxidation of all-cis 8,11,14-eicosatrienoate and arachidonate was discovered in the 60s.20 Detailed studies of the autoxidation of

polyunsaturated fatty acids were initiated in the 70s by several research groups,

disclosing more complex mixtures21-23 than those previously proposed. The further

demonstration of oxidation of membrane phospholipids24 (PL) led to a new fruitful era

with a continuous flow of considerable research devoted to chemistry, biochemistry and

medicine.

Lipid oxidation in diverse biological systems contributes to normal physiological

processes. Hydroperoxides derived from arachidonic acid serve as modulators of the

involved in the biosynthesis of prostaglandins and leukotrienes.25-28 Oxidized

phospholipids (oxPL) are suspected in the pathogenesis of atherosclerosis29-33 and several

chronic inflammatory diseases such as antiphospholipid antibody syndrome34,

rheumatoid arthritis35, inflammatory bowel disease36, and multiple sclerosis.37 Lipid

oxidation may also be involved in the immune response38 or apoptosis (programmed cell

death) 39, 40, and is increasingly believed to be associated with age-related diseases.41-43

Research on the chemistry of lipid oxidation and the reactions of lipid oxidation products led to the identification of some new biological effects such as the covalent adduction

5 that modifies DNA or protein function as well as the receptor-mediated responses

triggered by oxPL or lipid-based protein modifications that act as “oxidative

messengers”.39, 44 However, our understanding of the intrinsic role that lipid oxidation

plays in both physiological and pathological processes is still in its infancy. Research in

Dr. Salomon’s group aims to understand the biologically important chemistry of oxidized

lipids and to determine the extent and consequences of such chemistry in pathogenesis of human diseases.

1.3. 4-Hydroxy-2-nonenal and its protein adducts. An important effect of lipid

peroxidation is decreased membrane fluidity, which alters membrane integrity and can

significantly disrupt membrane-bound proteins. Moreover, the peroxidation of lipids

generates reactive aldehydes, that may act as mutagens45 or inactivate enzymes,46-48 or react with proteins and nucleic acids to form heterogeneous cross-links.49 One of these aldehydes, 4-hydroxy-2(E)-nonenal (HNE), has been investigated extensively since the report that it inhibits glucose 6-phosphatase and cytochrome P-450.47, 50 HNE is a product of oxidative fragmentation of arachidonic acid (AA) or linoleic acid (LA) phospholipid esters. It is highly cytotoxic to Ehrlich ascites tumour cells.51 It can cause lysis of

erythrocytes50 and arouse chemiluminescence and pentane production in isolated

hepatocytes.52

HNE is highly reactive towards the nucleophilic residues of proteins such as lysine,

histidine and cysteine and generates Michael and Schiff base adducts as well as a pyrrole

adduct.53, 54 HNE can also mediate the formation of lysine-lysine crosslinks.55

6 Among the numerous adducts that HNE forms with proteins, 2-pentylpyrrole (PP) adducts, that incorporate the ε-amino group of protein lysyl residues, have been cited as one example of an “advanced lipid peroxidation end product” (ALPE).54 Mean PP levels

in plasma from individuals with atherosclerosis are double those found in healthy

controls.56 HNE and its protein adducts are therefore considered as established markers

for oxidative stress.57

1.4. Levuglandins, isolevuglandins and their protein adducts. The discovery of

levuglandins was the culmination of a ten year effort to elucidate the chemistry of the

prostaglandin endoperoxide PGH2. PGH2 is the cyclooxygenase metabolite of

58 arachidonic acid (AA). Spontaneous rearrangements of PGH2 generate PGD2 and PGE2.

In addition, rearrangement of PGH2 in aqueous solution produces levulinaldehyde

23 derivatives with prostanoid side chains, namely levuglandins (LGs). Two LGs, LGD2

and LGE2, are generated (Scheme 1.1). Non-enzymatic (free radical-induced) oxidation

of arachidonyl phospholipids (AA-PL) gives rise to four structurally isomeric families of

phospholipid endoperoxide stereoisomers, that have been named isoprostanes (isoPs).

Rearrangement of these endoperoxides produces four LGE2 structural isomers, i. e.,

isoLGE2, iso[4]LGE2, iso[10]LGE2, iso[7]LGE2 and four corresponding LGD2 structural

isomers (Scheme 1.2).59

Levuglandins and isolevuglandins are biologically active. Guanosine triphosphate

(GTP)-induced assembly of bovine microtubule protein is inhibited by LGs and

60, 61 chymotrypsin-like proteasomal activity is effectively restrained by isoLGE2.

7 (CH ) COOH HO 2 3 C5H11 O (CH ) COOH AA 2 3 (CH2)3COOH

C H O2 cyclooxygenase 5 11 C5H11 O OH PGD 2 O OH LGD2 (CH2)3COOH + O O C H O 5 11 O (CH2)3COOH PGH OH 2 (CH2)3COOH

C5H11 C5H11 HO OH PGE2 O OH LGE2

Scheme 1.1. Cyclooxygenase oxidation of AA generates PGs and LGs via rearrangement of PGH2.

O 10 7 O O C H Arachidonyl O O 15 31 phosphatidylcholine C H O P N(CH3)3 ester 13 5 11 O O -7 H -10 H -10 H -13 H

OH OH (CH2)3CO2R 8 O (CH2)3CO2R O (CH2)3CO2R O (CH2)3CO2R O O O C5H11 O C5H11 O C5H11 9 OH iso[7]PGH2-PC iso[10]PGH2-PC OH iso[4]PGH2-PC isoPGH2-PC

O OH O O OH O

(CH2)3CO2R 8 (CH2)3CO2 (CH2)3CO2R (CH2)3CO2R R 12 C5H11 C5H11 C5H11 9 O O OH O O OH

iso[7]LGE2-PC iso[4]LGE2-PC iso[10]LGE2-PC isoLGE2-PC ++ + +

O OH O O OH O

(CH2)3CO2R 8 (CH2)3CO2 (CH2)3CO2R (CH2)3CO2R R 12 C5H11 C5H11 C5H11 9 O O OH O O OH

iso[7]LGD2-PC iso[4]LGD2-PC iso[10]LGD2-PC isoLGD2-PC Scheme 1.2. Free radical-induced oxidation of AA-PC produces isoLGs by rearrangement of isoP intermediates.

8 LGs and isoLGs covalently bind avidly with proteins within minutes due to their

reactive electrophilic γ-ketoaldehyde functionality.62 Moreover, protein-protein

cross-links and protein polymerization can occur in conjunction with the binding, and this

is believed to be associated with some pathological process, e. g., Alzheimer’s disease

(AD). Levels of levulglandinyl-lysine lactam adducts of proteins are elevated in AD hippocampi compared to normal.63

A Schiff base, formed initially, eventually is transformed into a lactam through an

intermediate pyrrole. The lactam is the primary end product form of LGE2-protein

adducts.64 The LG-protein Schiff base adduct can act as a reactive electrophile. It binds

with a primary amino group of another protein leading to a protein-protein crosslink. LGs

also cause DNA-protein cross-links (DPCs) which are repair-resistant and were shown to

be relevant to cell killing (Scheme 1.3).65

Levuglandin-protein adducts are detectable in vivo using polyclonal rabbit antibodies

and are useful as unambiguous markers of oxidative injury. The mean levels of

iso[4]LGE2 and iso[7]LGE2-protein adducts, as well as LGE2-protein adducts, are

significantly elevated in plasma from individuals with atherosclerosis and renal disease

compared to normal individuals.66, 67 In addition, oxidative stress induced by Candida

infection in mice leads to a 3.5-fold elevation in plasma levels of iso[4]LGE2-protein

adducts.68 In this thesis, more pathological diseases are explored to monitor the levels of

iso[4]LGE2-protein adducts in plasma and thereby to further understand the biological

significance of levulglandin chemistry.

9 bis-pyrrole adduct O R1 R1 protein N

levuglandin R2 H C R2 2 R1 O protein N

protein NH2 R2

H2O R1 R O 1 protein N protein N R2 R2 Schiff Base LG-pyrrole protein NH NH2 DNA 2 O2, . ROO

OH R1 R1 O O R1 N protein N protein protein N R2 R2 H H R2 NH NH O protein DNA hydroxylactam +

OH OH R R1 R1 1 protein protein N protein N N R R2 R2 2 protein NH DNA NH O lactam

protein-protein crosslink DNA-protein crosslink

Scheme 1.3. Formation of LG-protein adducts, protein-protein and DNA-protein

crosslinks.

1.5. Oxidatively truncated phospholipids and carboxyalkylpyrrole modifications of

proteins. Free radical induced oxidation of arachidonate (C20) and linoleate (C18) phospholipids generates truncated C5 or C9 ω-oxoalkanoate phospholipids, 2-oxovaleryl

(OV) or 9-oxononanoyl (ON) phospholipids, respectively, as well as C8 or C12

γ–hydroxyalkenal phospholipids, 5-hydroxy-8-oxo-6-octenoic acid (HOOA) or

10 9-hydroxy-12-oxododec-10-enoic acid (HODA), which are precursors of

carboxyalkylpyrrole derivatives with proteins (Scheme 1.4).69 Both OV

phosphatidylcholine (OV-PC), and HOOA-PC induce monocyte binding to endothelial

cells but inhibit lipopolysaccharide (LPS) induced E-selectin expression which is a major

endothelial adhesion molecule involved in neutrophil binding.70 Moreover, HOOA-PC

and HODA-PC and their protein modification products, 2-(ω-carboxypropyl) pyrroles

(CPPs) and 2-(ω-carboxyheptyl) pyrroles (CHPs), have been confirmed to be present in

human atherosclerotic lesions. HOOA-PC and HODA-PC also impair the proteolytic

degradation of internalized macromolecules by mouse peritoneal macrophages (MPMs)

and inhibit the activity of the lysosomal protease, cathepsin B, as well as block the

maturation of the fusion protein Rab5a.71 All these results implicate that the oxidatively truncated phospholipids may contribute to chronic inflammation such as atherosclerotic plaques associated with the accumulation of cholesterol esters and the formation of foam cells.

1.6. Carboxyethylpyrroles (CEPs) and their potential clinical applications. Oxidative cleavage of phospholipids containing docosahexaenoic acid (DHA) produces reactive electrophilic 4-hydroxy-7-oxohept-5-enoic acid (HOHA)-PL that convert the primary

amino group of protein lysyl residues into 2-(ω-carboxyethyl)pyrrole (CEP) derivatives

(Scheme 1.4). DHA is most abundant in the brain72 and retina, and is concentrated in the light-sensitive photoreceptor rod outer segment (ROS) membranes and the retinal pigment epithelium (RPE).73 The extremely high oxygen levels of the environment, the

11 exquisite sensitivity to being oxidized due to its six double bonds, make DHA especially

important in the pathological processes associated with retinal diseases.

Polyunsatuated Fatty Acid (PUFAs) Hydroxy-ω-oxoalkenoic Acid Carboxyalkylpyrrole

Protein OH C5H11 (CH2)7COOH O2 Protein linoleic acid (LA) O N(CH2)7COOH (CH2)7COOH C H (CH ) COOH 2 5 2 7 HODA CHP linolenic acid

C5H11 (CH2)3COOH γ-linolenic acid Protein OH O Protein 2 O N(CH2)3COOH C5H11 (CH2)3COOH 2 (CH2)3COOH arachidonic acid (AA) HOOA CPP

C2H5 (CH2)3COOH 3 eicosapentaenoic acid Protein OH O2 Protein O N(CH2)2COOH C2H5 (CH2)2COOH 4 (CH2)2COOH docosahexaenoic acid (DHA) HOHA CEP Scheme 1.4. Oxidation of polyunsaturated fatty acids generates hydroxyl-ω-oxoalkenoic acids, that react with proteins and form 2-(ω-carboxyalkyl)pyrroles.

There are promising results that implicate CEPs as clinically useful markers of

age-related macular degeneration (AMD). CEPs and autoantibodies against CEPs are

elevated in the blood of individuals with AMD.42 Western blot analysis of PAGE gels of

protein extracts from the choroid/RPE/ROS complex of AMD retinas revealed markedly

elevated levels of CEP immunoreactivity. Proteomic characterization of drusen, deposits

that are a risk factor for AMD and accumulate below the RPE on Bruch’s membrane, also showed the presence of CEPs.41 A recent report announced that CEPs could induce

angiogenesis by promoting sprouting of new vessels into the retina (choroidal

neovascularization) (Figure 1.2), and damaging the photoreceptor cells.43 CEP levels are

12 elevated by a factor of four in the retinas of rats exposed to intense light compared to those raised in the dark (unpublished data from Renganathan, K.).

A HSA B CEP-HSA

C Dipeptide D CEP-Dipeptide

Figure 1.2. CEP-HSA as well as dipeptide (Ac-gly-lys-OH) induces angiogenesis in a rat corneal micropocket assay. (A) HSA (1 μg), (B) CEP-HSA (1 μg), (C) dipeptide (41 ng),

(D) CEP-dipeptide (37 ng).

We recently conducted mouse model studies that showed retina degeneration, holes forming inside the retina which looks exactly like AMD symptoms, after one year of

CEP-mouse serum albumin (MSA) injection into mice (Figure 1.3).74 Moreover, a significant number of CEP-MSA specific interferon-γ (IFN-γ) and interleukin-2 (IL-2) producing T cells in lymph nodes and spleen of immunized mice as well as an increase of anti-CEP autoantibody titers were detected in immunized mice treated with CEP-MSA.75

13 These observations demonstrate that immune responses to a CEP adducted self-protein in

the retina can lead to the generation of an adaptive immune response that results in retinal

damage and possibly AMD. It is also suggested that an in vivo chemical modification

induced by oxidative stress in a specific organ can lead to autoimmunity.

Figure 1.3. Outer retinal pathology present in mice immunized with CEP-MSA (A-J).

Large arrows indicate several inflammatory cells immediately adjacent to the retinal pigment epithelium (RPE) or in the interphotoreceptor matrix (IPM). Large empty vacuoles are also evident (C-I). Some of which appear to be intracellular (I). In (J) the

RPE has degenerated (asterisks). Bar in lower right of (J) represent 25 μm.

In view of the abundance of DHA in brain tissues, it is presumed that CEP locates in the brain and its level might be related to neurological diseases. Immunohistochemical staining of cortical tissue from four autistic brains and five age-matched normal brains

14 using anti-CEP polyclonal antibody exhibited stained filaments in all of the autistic brains

while no staining in the control brains.76

In summary, there is increasing evidence that lipid oxidation products and lipid-derived

protein adducts are related to specific pathological processes. This thesis focuses on

understanding the role of oxidative injury in various diseases and clinically characterizing the disease-related levels of specific lipid oxidation products in tissues and plasma. These studies provide a foundation for the development of diagnostic methods and therapeutic interventions for diseases associated with oxidative injury.

Chapter 2 describes the development of an efficient synthesis of CEP derivatives such

as CEP-proteins, CEP-phosphatidylethanolamine (PE) and CEP biotinylated derivatives.

The synthesis of CEP-MSA enables the establishment of a mouse animal model to study

immune mechanisms that could lead to the development of AMD and could be used to

develop new therapies. The characterization of CEP-proteins was also accomplished

using tryptic digestion in conjunction with quadrupole time-of-flight (Q-TOF) mass

spectrometry and tandem mass spectrometric analysis.

Chapter 3 explores the contribution of oxidative injury to autism by monitoring levels

of iso[4]LGE2-protein adducts, CEP and corresponding autoantibody titers as well as nitrotyrosine in plasma from autistic patients and normal controls.

Chapter 4 assesses the effects of oral vitamin Eα supplementation on the levels of

pentosidine, iso[4]LGE2-, 2-pentylpyrrole-, and (E)-4-oxo-2-nonenal (ONE)-protein

adducts in hemodialysis patients.

15 Chapter 5 establishes methodology that was used to quantify CEP-PEs in lipid extracts

from bovine retina as well as from plasma samples of an AMD patient and a normal

control by using authentic standards. The formation of these adducts in vivo is predicted

to be associated with the pathogenesis of AMD.

Chapter 6 describes a pilot study of LPS-induced generation of LGE2 and iso[4]LGE2 protein modifications in mouse cornea model of keratitis.

Chapter 7 describes the total synthesis of a pyrazole isosteres of 2-pentylpyrroles.

16 1.7. References.

1. Harman, D., The aging process. Proc. Natl. Acad. Sci. USA 1981, 78, 7124-7128.

2. Harman, D., Aging: A theory based on free radical and radiation chemistry. J.

Gerontol. 1956, 2, 298-300.

3. Barja, G.; Cadenas, S.; Rojas, C.; Perez-Campo, R.; Lopez-Torres, M., Low mitochondrial free radical production per unit O2 consumption can explain the simultaneous presence of high longevity and high aerobic metabolic rate in birds. Free

Radic. Res. 1994, 21, (5), 317-27.

4. Ku, H. H.; Brunk, U. T.; Sohal, R. S., Relationship between mitochondrial superoxide and production and longevity of mammalian species. Free

Radic. Biol. Med. 1993, 15, (6), 621-7.

5. Sohal, R. S.; Ku, H. H.; Agarwal, S., Biochemical correlates of longevity in two closely related rodent species. Biochem. Biophys. Res. Commun. 1993, 196, (1), 7-11.

6. Sohal, R. S.; Sohal, B. H.; Orr, W. C., Mitochondrial superoxide and hydrogen peroxide generation, protein oxidative damage, and longevity in different species of flies.

Free Radic. Biol. Med. 1995, 19, (4), 499-504.

7. Finkel, T.; Holbrook, N. J., Oxidants, oxidative stress and the biology of ageing.

Nature 2000, 408, (6809), 239-47.

8. O'Donnell, V. B., Free Radicals and Lipid Signaling in Endothelial Cells.

Antioxidants & Redox Signaling 2003, 5, 195 -203

9. Kalyanaraman, B.; and Gutterman, D. D., Prologue: Vascular effects of free radicals

17 Am. J. Physiol. Heart Circ. Physiol. 2003, 285, H2253-H2254.

10. Nishikawa, T.; Edelstein, D.; Du, X. L.; Yamagishi, S.; Matsumura, T.; Kaneda, Y.;

Yorek, M. A.; Beebe, D.; Oates, P. J.; Hammes, H. P.; Giardino, I.; Brownlee, M.,

Normalizing mitochondrial superoxide production blocks three pathways of

hyperglycaemic damage. Nature 2000, 404, (6779), 787-90.

11. Nemoto, S.; Takeda, K.; Yu, Z. X.; Ferrans, V. J.; Finkel, T., Role for mitochondrial

oxidants as regulators of cellular metabolism. Mol. Cell. Biol. 2000, 20, (19), 7311-8.

12. Finkel, T., Oxygen radicals and signaling. Curr. Opin. Cell Biol. 1998, 10, (2),

248-53.

13. See: "Free radical introduction" at

http://www.exrx.net/Nutrition/Antioxidants/Introduction.html.

14. Knight, J. A., Review: Free radicals, antioxidants, and the immune system Ann. Clini.

Lab. Sci. 2000, 30, 145-158.

15. Dekkers, J. C.; Van Doornen, L. J.; and Kemper, H. C. G., The Role of Antioxidant

Vitamins and Enzymes in the Prevention of Exercise-Induced Muscle Damage. Sports

Med. 1996, 21, 213-238.

16. Kaczmarski, M., J. Wojicicki, L. Samochowiee, T. Dutkiewicz, and Z. Sych., The

influence of exogenous antioxidants and physical exercise on some parameters associated with production and removal of free radicals. Pharmazie. 1999, 54, 303-306.

17. See: "Introduction to lipid peroxidation" at

http://www.cyberlipid.org/perox/oxid0002.htm.

18 18. Criegee, R.; Pilz, H.; Flygare, H., Chem. Ber. 1939, 72, 1799.

19. Bolland, J. L., Kinetics of olefin oxidation. Q. Rev. Chem. Soc. 1949, 3, 1.

20. Nugteren, D. H.; Vonkeman, H.; Dorp, D. A., Non-enzymic conversion of all-cis 8,

11, 14-eicosatrienoic acid into prostaglandin E1. Rec. Trav. Chim. 1967, 86, 1237-1245.

21. Porter, N. A.; Weber, B. A.; Weenen, H.; Khan, J. A., Autoxidation of

polyunsaturated lipids. Factors controlling the stereochemistry of product hydroperoxides

J. Am. Chem. Soc. 1980, 102, 5597-5601.

22. Salomon, M. F.; Salomon, R. G., 2,3-Dioxabicyclo[2.2.1]heptane. The strained cyclic

peroxide nucleus of prostaglandin endoperoxides. J. Am. Chem. Soc. 1977, 99, 3501.

23. Salomon, R. G.; Miller, D. B.; Zagorski, M. G.; Coughlin, D. J., Solvent induced

fragmentation of prostaglandin endoperoxides. New aldehyde products from PGH2 and a

novel intramolecular 1,2-hydride shift during endoperoxide fragmentation in aqueous solution. J. Am. Chem. Soc. 1984, 106, 6049-6060.

24. Porter, N. A.; Wolf, R. A.; and Weenen, H., The free radical oxidation of

polyunsaturated lecithins Lipids 1980, 15, 163-167.

25. Hamberg, M.; Samuelsson, B., Prostaglandin endoperoxides. Novel transformations

of arachidonic acid in human platelets. Proc. Natl. Acad. Sci. U. S. A. 1974, 71, (9),

3400-4.

26. Nugteren, D. H., Arachidonate lipoxygenase in blood platelets. Biochim. Biophys.

Acta 1975, 380, (2), 299-307.

27. Borgeat, P.; Samuelsson, B., Transformation of arachidonic acid by rabbit

19 polymorphonuclear leukocytes. Formation of a novel dihydroxyeicosatetraenoic acid. J.

Biol. Chem. 1979, 254, (8), 2643-6.

28. Borgeat, P.; Samuelsson, B., Arachidonic acid metabolism in polymorphonuclear leukocytes: effects of ionophore A23187. Proc. Natl. Acad. Sci. U. S. A. 1979, 76, (5),

2148-52.

29. Steinberg, D.; Parthasarathy, S.; Carew, T. E.; Khoo, J. C.; Witztum, J. L., Beyond cholesterol. Modifications of low-density lipoprotein that increase its atherogenicity. N.

Engl. J. Med. 1989, 320, (14), 915-24.

30. Steinberg, D., J. Biol. Chem. 1997, 272, 20963-20966.

31. Siqueira, A. F.; Abdalla, D. S.; Ferreira, S. R., [LDL: from metabolic syndrome to instability of the atherosclerotic plaque]. Arq. Bras. Endocrinol. Metabol. 2006, 50, (2),

334-43.

32. Spiteller, G., The relation of lipid peroxidation processes with atherogenesis: a new theory on atherogenesis. Mol. Nutr. Food Res. 2005, 49, (11), 999-1013.

33. Yla-Herttuala, S.; Palinski, W.; Rosenfeld, M. E.; Parthasarathy, S.; Carew, T. E.;

Butler, S.; Witztum, J. L.; Steinberg, D., Evidence for the presence of oxidatively modified low density lipoprotein in atherosclerotic lesions of rabbit and man. J. Clin.

Invest. 1989, 84, (4), 1086-95.

34. Horkko, S.; Miller, E.; Dudl, E.; Reaven, P.; Curtiss, L. K.; Zvaifler, N. J.; Terkeltaub,

R.; Pierangeli, S. S.; Branch, D. W.; Palinski, W.; Witztum, J. L., Antiphospholipid antibodies are directed against epitopes of oxidized phospholipids. Recognition of

20 cardiolipin by monoclonal antibodies to epitopes of oxidized low density lipoprotein. J.

Clin. Invest. 1996, 98, (3), 815-25.

35. Mapp, P. I.; Grootveld, M. C.; Blake, D. R., Hypoxia, oxidative stress and rheumatoid arthritis. Br. Med. Bull. 1995, 51, (2), 419-36.

36. Grisham, M. B., Oxidants and free radicals in inflammatory bowel disease. Lancet

1994, 344, (8926), 859-61.

37. Toshniwal, P. K.; Zarling, E. J., Evidence for increased lipid peroxidation in multiple sclerosis. Neurochem. Res. 1992, 17, (2), 205-7.

38. McMurray, H. F.; Parthasarathy, S.; Steinberg, D., Oxidatively modified low density lipoprotein is a chemoattractant for human T lymphocytes. J. Clin. Invest. 1993, 92, (2),

1004-8.

39. Greenberg, M. E.; Sun, M.; Zhang, R.; Febbraio, M.; Silverstein, R.; Hazen, S. L.,

Oxidized phosphatidylserine-CD36 interactions play an essential role in macrophage-dependent phagocytosis of apoptotic cells. J. Exp. Med. 2006, 203, (12),

2613-25.

40. Okura, Y.; Brink, M.; Itabe, H.; Scheidegger, K. J.; Kalangos, A.; Delafontaine, P.,

Circulation 2000, 102, 2680-2686.

41. Crabb, J. W.; Miyagi, M.; Gu, X.; Shadrach, K.; West, K. A.; Sakaguchi, H.; Kamei,

M.; Hasan, A.; Yan, L.; Rayborn, M. E.; Salomon, R. G.; Hollyfield, J. G., Drusen proteome analysis: an approach to the etiology of age-related macular degeneration. Proc.

Natl. Acad. Sci. U. S. A. 2002, 99, (23), 14682-7.

21 42. Gu, X.; Meer, S. G.; Miyagi, M.; Rayborn, M. E.; Hollyfield, J. G.; Crabb, J. W.;

Salomon, R. G., Carboxyethylpyrrole protein adducts and autoantibodies, biomarkers for age-related macular degeneration. J. Biol. Chem. 2003, 278, (43), 42027-35.

43. Ebrahem, Q.; Renganathan, K.; Sears, J.; Vasanji, A.; Gu, X.; Lu, L.; Salomon, R. G.;

Crabb, J. W.; Anand-Apte, B., Carboxyethylpyrrole oxidative protein modifications

stimulate neovascularization: Implications for age-related macular degeneration. Proc.

Natl. Acad. Sci. U. S. A. 2006, 103, (36), 13480-4.

44. Sambrano, G. R.; Parthasarathy, S.; Steinberg, D., Recognition of oxidatively

damaged erythrocytes by a macrophage receptor with specificity for oxidized low density

lipoprotein. Proc. Natl. Acad. Sci. U. S. A. 1994, 91, (8), 3265-9.

45. Marnett, L. J.; Hurd, H. K.; Hollstein, M. C.; Levin, D. E.; Esterbauer, H.; Ames, B.

N., Naturally occurring carbonyl compounds are mutagens in Salmonella tester strain

TA104. Mutat. Res. 1985, 148, (1-2), 25-34.

46. Chen, J. J.; Yu, B. P., Alterations in mitochondrial membrane fluidity by lipid

peroxidation products. Free Radic. Biol. Med. 1994, 17, (5), 411-8.

47. Szweda, L. I.; Uchida, K.; Tsai, L.; Stadtman, E. R., Inactivation of

glucose-6-phosphate dehydrogenase by 4-hydroxy-2-nonenal. Selective modification of

an active-site lysine. J. Biol. Chem. 1993, 268, (5), 3342-7.

48. Uchida, K.; Szweda, L. I.; Chae, H. Z.; Stadtman, E. R., Immunochemical detection

of 4-hydroxynonenal protein adducts in oxidized hepatocytes. Proc. Natl. Acad. Sci. U. S.

A. 1993, 90, (18), 8742-6.

22 49. Chio, K. S.; Tappel, A. L., Synthesis and characterization of the fluorescent products derived from malonaldehyde and amino acids. Biochemistry 1969, 8, (7), 2821-6.

50. Benedetti, A.; Comporti, M.; & Esterbauer, H., Biochim. Biophys. Acta 1980, 620,

281-296.

51. Schauenstein, E.; Esterbauer, H.; & Zollner, H., Aldehydes in Biological Systems,

1977, Pion, London.

52. Cadenas, E.; Muller, A.; Brigelius, R.; Esterbauer, H.; Sies, H., Biochem. J. 1983,

214, 479-487.

53. Uchida, K., Amino Acids 2003, 25, 249-257.

54. Sayre, L. M.; Sha, W.; Xu, G.; Kaur, K.; Nadkarni, D.; Subbanagounder, G.; Salomon,

R. G., Immunochemical evidence supporting 2-pentylpyrrole formation on proteins exposed to 4-hydroxy-2-nonenal. Chem. Res. Toxicol. 1996, 9, (7), 1194-201.

55. Xu, G.; Sayre, L. M., Chem. Res. Toxicol. 1998, 11, 247-251.

56. Salomon, R. G.; Kaur, K.; Podrez, E.; Hoff, H. F.; Krushinsky, A. V.; Sayre, L. M.,

HNE-derived 2-pentylpyrroles are generated during oxidation of LDL, are more prevalent in blood plasma from patients with renal disease or atherosclerosis, and are present in atherosclerotic plaques. Chem. Res. Toxicol. 2000, 13, (7), 557-64.

57. Zarkovic, N., 4-hydroxynonenal as a bioactive marker of pathophysiological processes. Mol. Aspects Med. 2003, 24, (4-5), 281-91.

58. Hamberg, M.; Samuelsson, B., Detection and isolation of an endoperoxide intermediate in prostaglandin biosynthesis. Proc. Natl. Acad. Sci. U. S. A. 1973, 70, (3),

23 899-903.

59. Salomon, R. G.; Subbanagounder, G.; Singh, U.; O'Neil, J.; Hoff, H. F., Oxidation of

low-density lipoproteins produces levuglandin-protein adducts. Chem. Res. Toxicol. 1997,

10, (7), 750-9.

60. Murthi, K. K.; Salomon, R. G.; Sternlicht, H., Levuglandin E2 inhibits mitosis and

microtubule assembly. Prostaglandins 1990, 39, (6), 611-22.

61. Davies, S. S.; Amarnath, V.; Montine, K. S.; Bernoud-Hubac, N.; Boutaud, O.;

Montine, T. J.; Roberts, L. J., 2nd, Effects of reactive gamma-ketoaldehydes formed by

the isoprostane pathway (isoketals) and cyclooxygenase pathway (levuglandins) on

proteasome function. Faseb J. 2002, 16, (7), 715-7.

62. Salomon, R. G.; Jirousek, M. R.; Ghosh, S.; Sharma, R. B., Prostaglandin

endoperoxides 21. Covalent binding of levuglandin E2 with proteins. Prostaglandins

1987, 34, (5), 643-56.

63. Zagol-Ikapitte, I.; Masterson, T. S.; Amarnath, V.; Montine, T. J.; Andreasson, K. I.;

Boutaud, O.; Oates, J. A., Prostaglandin H(2)-derived adducts of proteins correlate with

Alzheimer's disease severity. J. Neurochem. 2005, 94, (4), 1140-5.

64. Boutaud, O.; Brame, C. J.; Salomon, R. G.; Roberts, L. J., 2nd; Oates, J. A.,

Characterization of the lysyl adducts formed from prostaglandin H2 via the levuglandin

pathway. Biochemistry 1999, 38, (29), 9389-96.

65. Murthi, K. K.; Friedman, L. R.; Oleinick, N. L.; Salomon, R. G., Formation of

DNA-protein cross-links in mammalian cells by levuglandin E2. Biochemistry 1993, 32,

24 (15), 4090-7.

66. Salomon, R. G.; Batyreva, E.; Kaur, K.; Sprecher, D. L.; Schreiber, M. J.; Crabb, J.

W.; Penn, M. S.; DiCorletoe, A. M.; Hazen, S. L.; Podrez, E. A., Isolevuglandin-protein

adducts in humans: products of free radical-induced lipid oxidation through the isoprostane pathway. Biochim. Biophys. Acta 2000, 1485, (2-3), 225-35.

67. Salomon, R. G.; Kaur, K.; Batyreva, E., Isolevuglandin-protein adducts in oxidized low density lipoprotein and human plasma: a strong connection with cardiovascular disease. Trends Cardiovasc. Med. 2000, 10, (2), 53-9.

68. Poliakov, E.; Brennan, M. L.; Macpherson, J.; Zhang, R.; Sha, W.; Narine, L.;

Salomon, R. G.; Hazen, S. L., Isolevuglandins, a novel class of isoprostenoid derivatives,

function as integrated sensors of oxidant stress and are generated by myeloperoxidase in

vivo. Faseb J. 2003, 17, (15), 2209-20.

69. Kaur, K.; Salomon, R. G.; O'Neil, J.; Hoff, H. F., (Carboxyalkyl)pyrroles in human

plasma and oxidized low-density lipoproteins. Chem. Res. Toxicol. 1997, 10, (12),

1387-96.

70. Subbanagounder, G.; Deng, Y.; Borromeo, C.; Dooley, A. N.; Berliner, J. A.; Salomon,

R. G., Hydroxy alkenal phospholipids regulate inflammatory functions of endothelial

cells. Vascul. Pharmacol. 2002, 38, (4), 201-9.

71. Hoff, H. F.; O'Neil, J.; Wu, Z.; Hoppe, G.; Salomon, R. L., Phospholipid

hydroxyalkenals: biological and chemical properties of specific oxidized lipids present in

atherosclerotic lesions. Arterioscler. Thromb. Vasc. Biol. 2003, 23, (2), 275-82.

25 72. Skinner, E. R.; Watt, C.; Besson, J. A.; Best, P. V., Differences in the fatty acid composition of the grey and white matter of different regions of the brains of patients with Alzheimer's disease and control subjects. Brain 1993, 116 ( Pt 3), 717-25.

73. Wang, N.; Anderson, R. E., Enrichment of polyunsaturated fatty acids from rat retinal pigment epithelium to rod outer segments. Curr. Eye Res. 1992, 11, (8), 783-91.

74. Hollyfield, J. G.; Rayborn, M. E.; Yang, X.; Ufret, R.; Lu, L.; Yu, M.; Shadrach, K. G.;

Peachey, N. S.; Salomon, R. G.; Perez, V. L., Identification of an Inflammatory Signal

From the Outer Retina Causing Age-Related Macular Degeneration. 2007 ARVO Annual

Meeting, May 6-10, 2007, Fort Lauderdale, FL, U. S. A.

75. Perez, V. L.; Yang, X.; Raybourn, M. E.; Ufret, R.; Yu, M.; Lu, L.; Salomon, R. G.;

Peachey, N. S.; Hollyfield, J. G., Auto-Immune Responses to Oxidatively Altered Self

Proteins in the Retina Lead to the Development of Retinal Degeneration. 2007 ARVO

Annual Meeting. May 6-10, 2007, Fort Lauderdale, FL, U. S. A.

76. Evans, T.; Siedlak, S.; Lu, L.; Fu, X.; Wang, Z.; McGinnis, W.; Fakhoury, E.;

Castellani, R.; Hazen, S. L.; Walsh, W. J.; Salomon, R. G.; Smith, M. A.; Perry, G.; Zhu,

X., The Autistic Phenotype Exhibits a Remarkably Localized Modification of Brain

Protein by Products of Free Radical-Induced Lipid Oxidation. Am. J. Biochem.

Biotechnol. 2007, Submitted.

26

Chapter 2

Syntheses and Characterization of Carboxyethylpyrroles

27 2.1. Background

Although scarce in most human tissues, docosahexaenoic acid (DHA) is essential for

the growth, functional development and maintenance of the brain.1 Phospholipids

containing DHA are major structural elements of photoreceptor cell membranes in the

retina.2, 3 Owing to the presence of the six homoconjugated C=C bonds in DHA, it is exquisitely sensitive to oxidative damage.4 Oxidative cleavage of retinal phospholipids

containing DHA produces reactive electrophilic 4-hydroxy-7-oxohept-5-enoates that

convert the primary amino group of protein lysyl residues into 2-(ω-carboxyethyl)pyrrole

(CEP) derivatives. CEPs are especially abundant in the retinas of individuals with age-

related macular degeneration (AMD), a slow, progressive disease of the eye5 that is the

major cause of untreatable loss of vision among the elderly in developed countries.6, 7

Roughly 11 % of people in the United States have AMD, and owing to increases in

human life span, AMD is expected to nearly double in the next 25 years.8, 9 Proteomic

characterization of drusen, deposits that accumulate in the back of the retina, revealed

that CEP adducts are associated with drusen proteins.10 CEPs and autoantibodies against

CEPs are elevated in the blood of individuals with AMD.11 CEPs are not simply benign

markers of oxidative damage. We recently reported that CEPs promote the growth of

capillaries into the retina (choroidal neovascularization), and destruction of the

photoreceptor cells.12 Such neovascularization is responsible for 90 % of the loss of

vision associated with AMD.

This chapter describes the development of an efficient synthesis of CEPs that will

enable (1) their use as biomarkers for clinical prognosis of AMD, (2) their use as antigens

to test the hypothesis that immune responses against CEP-protein adducts generated in

the retina contribute to the pathogenesis of AMD through the activation of B- and T-cells,

28 (3) their use as “bait” to capture human ScFv antibodies from a phage display library, and

(4) for studies on the stimulation of angiogenesis by CEPs and its inhibition. The key

finding of this study is that a 9-fluorenylmethyl ester of 4,7-dioxoheptanoic acid reacts

with primary amines to provide esters of CEPs that can be deprotected with DBU without

causing protein denaturation. The introduction of multiple CEPs into proteins is readily

achieved using this strategy. In addition, the preparation of CEPs tethered to proteins

through an ω-amino hexanoate linker and their strong binding with anti-CEP antibodies is described. Characterization of the CEP adducts generated on the lysyl primary amino groups of proteins was achieved by quadrupole time-of-flight (Q-TOF) mass spectrometry and MS/MS analysis.

29 2.2. Results and Discussion

2.2.1. Paal-Knoor synthesis using 4,7-dioxoheptanoic acid is ineffective for the

preparation of CEPs. The reaction of γ-ketoaldehydes with primary amines, the Paal-

Knoor synthesis, is generally an efficient method for the preparation of pyrroles.13 This reaction was successfully applied to the generation of carboxyheptylpyrrole and carboxypropylpyrrole derivatives through the reactions of 9,12-dioxododecanoic or 5,8- dioxooctanoic acid with proteins. However, attempts at preparing the corresponding carboxyethylpyrrole derivatives of proteins by treatment with 4,7-dioxoheptanoic acid

(DOHA) generally caused precipitation, and in the few instances in which precipitation did not occur, the ratio of pyrrole to protein, e.g. 1.6:1 for human serum albumin,11 was

much lower than were obtained previously for the longer chain carboxyalkylpyrroles.14

Another distinguishing feature of DOHA was the nearly complete absence of a signal for the aldehydic hydrogen in its 1H NMR spectrum. We postulated that the unusual 1H

NMR spectrum and abberant reactivity of DOHA is a consequence of the proximity of the carboxyl group to the γ-ketoaldehyde array, and that DOHA exists in equilibrium with a spiroacylal hemiacetal (Scheme 2.1). To obviate complications engendered by the carboxyl group, we sought a masked derivative that could be deprotected under conditions that would not lead to denaturation and consequent precipitation of proteins.

This excluded acidic conditions. Therefore, we opted for a 9-fluorenylmethyl ester.

OH O N O O OH Protein CEP O DOHA NH HO 2 O Protein O O

Scheme 2.1. DOHA exists in equilibrium with a spiroacylal hemiacetal.

30 2.2.2. Synthesis of a 9-fluorenylmethyl (Fm) ester of DOHA. Coupling of 3-

carbomethoxypropionyl chloride with the Grignard reagent derived from 2-(2-

bromoethyl)-1,3-dioxolane provides access to the methyl ester 2.1 (Scheme 2.2). The

desired 9H-fluoren-9-ylmethyl ester 4,7-dioxo-heptanoic acid (DOHA-Fm, 2.4) was then obtained through saponification to afford the carboxylic acid 2.2, and esterificaton with

9-fluorenylmethanol, followed by hydrolysis of ethylene ketal in 2.3.

O O O Br OR 1) Mg, THF O O 2) ClCO(CH ) COOMe O 2 2 2.1, R = CH (60%) THF, -78 °C 3 NaOH, H O/MeOH/THF 2.2, R = H (90%) 2 FmOH, DCC, DMAP, CH Cl 2.3, R = Fm (95%) 2 2

O 88% O O HOAc:H2O (3:1, v/v) O 50 °C DOHA-Fm (2.4)

Scheme 2.2. Synthesis of 9H-fluoren-9-ylmethyl ester 4,7-dioxo-heptanoic acid (DOHA-

Fm, 2.4).

2.2.3. Syntheses of CEP-peptide and CEP-protein adducts by Paal-Knoor synthesis

with DOHA-Fm. Paal-Knoor synthesis of the Fm ester 2.5 of a CEP dipeptide was

O O O O H H N N N OMe N OMe H H O 80% O DOHA-Fm methanol

NH2 N COOR

Ac-Gly-Lys-OMe 2.5, R = Fm DBU,THF 2.6, R = H 86%

Scheme 2.3. Synthesis of CEP-dipeptide.

31 readily achieved in 80 % yield by the condensation of methyl 6-amino-2-((2-

acetylamino)acetyl)amino)hexanoate (Ac-Gly-Lys-OMe) with 1 equivalent of DOHA-

Fm (Scheme 2.3). For peptide synthesis, the deprotection of 9-fluorenylmethyl esters is

normally accomplished with piperidine in DMF. Piperidine serves both as a base to

fragment the Fm group and as a scavenger to trap the dibenzofulvene (DBF) released.15

The removal of an Fm group from an ester bound to a protein has not been reported.

Piperidine was unsatisfactory, vide infra. This was readily determined by the persistence of a UV absorbtion at 265 nm that is characteristic of the flourene group. Therefore, we examined the efficacy of a stronger base. 1,8-Diazabicyclo[5.4.0] undec-7-ene (DBU) successfully removed all Fm groups from protein adducts, vide infra, and this reagent was also applied to deprotection of the dipeptide Fm ester 2.5. Removal of the Fm protecting group by treatment of 2.5 with DBU in THF solution delivered the CEP dipeptide 2-(2-acetylamino-acetylamino)-6-[2-(2-carboxy-ethyl)-pyrrol-1-yl]-hexanoic acid methyl ester (2.6) in 86 % yield.

CEP-modified proteins are needed for immunoassays that measure levels of CEPs or anti-CEP autoantibodies in vivo. We also required CEP-protein adducts as antigens to immunize animals as models to test the hypothesis that immune responses against CEP- protein adducts generated in the retina contribute to the pathogenesis of AMD. We now find that the preparation of CEP-protein adducts can be readily accomplished by incubation of DOHA-Fm (2.4) with protein in 30 % DMF/phosphate-buffered saline

(PBS) solution for 5 days at 37 ˚C followed by deprotection by addition of DBU to the reaction mixture and stirring for an additional 9 h. One equivalent of DOHA-Fm was used for each lysine group present in a particular protein (Scheme 2.4). Proteins used for making CEP adducts were: human serum albumin (HSA), mouse serum albumin (MSA),

32 chicken egg albumin (CEO), rabbit myoglobin and glycerol-3-phosphate dehydrogenase

(GPDH). Low molecular weight contaminants were removed by dialysis (Mr cutoff

14000) of the reaction mixture against 20% DMF in 10 mM PBS. An especially

important feature of the use of Fm esters of DOHA is the ease with which residual Fm

groups can be detected and their complete removal assured by UV spectroscopy, i. e.,

absorption at 265 nm. The final protein concentration was determined by the Pierce

bicinchoninic acid (BCA) protein assay16 or Bio-rad protein assay.17 The pyrrole concentration was determined by the generation of a characteristic chromophore through reaction with 4-(dimethylamino)benzaldehyde, the Ehrlich reagent,18 using the CEP

dipeptide 2.6 as a quantitative standard. In contrast with the preparation of CEP-HSA by direct treatment with DOHA, that delivered a pyrrole to protein ratio of 1.6:1 for CEP-

HSA,11 the new synthetic method using DOHA-Fm provided CEP-HSA (2.7) with a

much higher pyrrole to protein ratio, 7.6 ± 1.1 to 1, and provided CEP-MSA (2.8) with a

pyrrole to protein ratio of 5.2 ± 1.0 to 1. Further characterization of the CEP adducts

generated on the primary amino groups of protein lysyl residues was achieved by QTOF

mass spectrometry and MS/MS analysis.

NH2 O N protein DOHA-Fm (2.4) O DMF, PBS protein protein = HSA protein = MSA protein = CEO protein = myoglobin 2.7, protein = HSA protein = GPDH COOH 2.8, protein = MSA N DBU,DMF, PBS 2.9, protein = CEO protein 2.10, protein = myoglobin 2.11, protein = GPDH

Scheme 2.4. Syntheses of CEP protein adducts.

33 2.2.4. CEP linked to proteins with an ω-aminohexanoyl tether. Direct coupling of

DOHA-Fm to proteins results in a high yield of CEP modifications of lysyl residues. As an alternative approach for anchoring CEP haptens to proteins for use as coating agents to capture anti-CEP antibodies, we examined the utility of CEPs anchored to proteins through hexanoyl amides of protein lysyl residues. An Fm masked 2-carboxyethylpyrrole

2.12 was generated through the reaction of DOHA-Fm with 6-aminocaproic acid. After purification, 2.12 was activated by conversion into an N-hydroxysuccinimide ester 2.13

(CEPFmSu). Incubation of the active ester 2.13 with bovine serum albumin (BSA)

( CEPFmSu : Lys = 1.5 :1, mol/mol), followed by deprotection in situ by addition of

DBU to the reaction mixture, delivered a 6-(2-carboxyethyl-1-pyrrolyl)hexanoyl amide derivative of BSA, CEPH-BSA (2.14, Scheme 2.5). Low molecular weight impurities were readily removed by dialysis (Mr cutoff 14000) with 20% DMF in 10 mM PBS and then with 10 mM PBS. The protein concentration was determined by a modified Lowry protein assay19 using the Lowry protein assay reagent and Folin-Ciocalteu reagent. The pyrrole concentration was determined using an Ehrlich assay. The pyrrole to BSA ratio in

CEPH-BSA (2.14) was 5.4 ± 0.9 to 1. CEP linked to Mouse albumin, chick albumin and

GPDH with this tether was also prepared.

34 O HOOC(CH ) NH 90% 2 5 2 HOOC(CH2)5N 80% N OOC(CH2)5N + HOSu H O 2 DCC DOHA-Fm (2.4) MeOH O 2.12 COO-Fm 2.13 CH2Cl2 COO-Fm DMF, PBS O O NH2 HN (CH ) N HN (CH2)5N 2 5 DBU protein protein protein DMF protein = BSA PBS COOH protein = MSA COO-Fm protein = CEO 2.14, CEPH-BSA, protein = BSA, protein = GPDH 2.15, CEPH-MSA, protein = MSA 2.16, CEPH-CEO, protein = CEO 2.17, CEPH-GPDH, protein = GPDH Scheme 2.5. Syntheses of CEPs bound to proteins through a tether.

The antibody binding affinity of CEPH-BSA (2.14) was determined by competitive

enzyme-linked immunosorbent assay (ELISA)20 using an anti-CEP-KLH polyclonal

antibody (Figure 2.1). CEP-HSA was used as a coating agent and standard whose binding was inhibited by CEPH-BSA. The IC50 of CEPH-BSA (1.93 pmol/mL) is lower than the

IC50 of CEP-HSA (3.02 pmol/mL) indicating that this preparation of CEPH-BSA has a slightly higher affinity than CEP-HSA for binding anti-CEP-KLH antibody.

To optimize the coupling reaction conditions, we used different protein concentrations

(1 mg/mL, 2.6 mg/mL, 3 mg/mL, 4 mg/mL, and 10 mg/mL), and coupled the protein with different CEPFmSu:Lys mole ratios (0.5:1, 1:1, 1.5:1). The pyrrole concentration

(relative to protein) of different coupling reactions was compared after normalizing the final protein concentration to 1 mg/mL (Table 2.1). The best coupling was achieved by incubating 3 mg/mL BSA with CEPFmSu:Lys 1.5:1 in 15% DMF.

35 120

100

80

60

IC50 = 3.02 40

20 IC50 = 1.93 Absorbance (%max)

0

0.01 0.1 1 10 100 1000 Inhibitor (pmol/ml)

Figure 2.1. Inhibition curves showing cross-reactivity of the anti-CEP-KLH antibody for CEP-HSA (▲) and CEPH-BSA (●) against CEP-HSA as coating agent.

Table 2.1. Pyrrole concentration (μM) generated in 1 mg/mL CEPH-BSA prepared by coupling various initial protein concentrations with various CEPFmSu/Lys ratios.

Protein Conc. 1 mg/ml 2.6 mg/ml 3 mg/ml 4 mg/mL 10 mg/mL CEPFmSu:Lys 0.5:1 20 25 35 N/A N/A

1:1 28 N/A 40 31 N/A

1.5:1 30 34 42 33 precipitate

2.2.5. Syntheses of biotinylated CEP derivatives. We want to explore the utility of monoclonal scFv antibodies (vide infra) as competitive inhibitors of the choroidal neovascularization that is promoted by CEPs. Immunoglobins and B-cells bind divalently through their variable (Fv) regions with haptens such as CEP. This can cause aggregation

36 of proteins and/or cells displaying CEP modifications, and can activate B-cells.

Immunoglobins also possess a constant (Fc) effector region that activates immune responses. In contrast, monoclonal scFv antibodies are monovalent constructs that contain only a single Fv binding region and no constant Fc region, and are therefore not expected to cause protein aggregation and deposition. Furthermore, monoclonal scFv antibodies are likely to favor penetration of the internal limiting membrane and access the subretinal space when injected intravitreously because of their relatively low molecular weight.21 Therefore, we anticipate that a monoclonal anti-CEP scFv antibody will be useful as a competitive inhibitor of CEP induced neovascularization in the eye.

Specific human scFv antibodies can be selected from engineered libraries of phage that display the antibody protein on their exterior. Selection is accomplished by using the corresponding hapten as “bait” to catch phage that produce (with the help of E. coli) and display antigen specific scFv on their exterior. One approach involves anchoring the hapten, through a biotin linker, to streptavidin-coated magnetic beads. For this purpose we prepared the biotinylated CEP Fm ester (2.20) from DOHA-Fm and 4-amino butylbiotin (Scheme 2.6). Deprotection of the intermediate 3-(1-[4-[5-(2-Oxo-hexahydro- thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-butyl]-1H-pyrrol-2-yl)-propionic acid 9H- fluoren-9-ylmethyl ester 2.20 by treatment with DBU in THF generated 3-(1-[4-[5-(2-

Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-butyl]-1H-pyrrol-2-yl)- propionic acid (2.21).

37 O O H N O-t-Bu NH NH 80% NH NH H2N(CH2)4 O O O H (CH2)4 N O-t-Bu S (CH2)4 OPhNO S (CH2)4 N 2 H 2.18 O O O 90% 84% ROOC NH NH NH NH CF3COOH O DOHA-Fm MeOH O CH2Cl2 (CH ) NH (CH ) N 2 4 2 (CH2)4 N S 2 4 S (CH2)4 N H H 2.19 2.20, R = Fm DBU, THF 2.21, R = H 85% Scheme 2.6. Synthesis of biotinylated CEP derivative 2.21.

All of the functionality in biotinylated CEP derivative 2.21 survived treatment with

sodium hydroxide ethanol solution. This observation suggested the feasibility of simpler

synthesis for preparing CEP derivatives of substrates that are stable to strong base. Thus,

we prepared 3-(1-[4-[5-(2-oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino] -

hexyl]-1H-pyrrol-2-yl)-propionic acid (2.25) by reacting 4,7-dioxo-heptanoic acid methyl

ester (DOHA-Me, 2.22) with biotinyl-1,6-diaminohexane (2.23) followed by hydrolysis

of the methyl ester 2.24 with ethanolic sodium hydroxide (Scheme 2.7).

O O OMe O O 87% OMe H O O 2.1 O 2 HOAc 2.22 O O O 90% NH NH O NH NH O H2N(CH2)6NH2 OC H NO pyridine 6 4 2 NH(CH2)6NH2 S H2O S O 2.23

NH NH 2.22 75% O + MeOH N 2.23 NH(CH2)6 S O 2.24, R = CH 3 NaOH, EtOH 2.25, R = H OR 84%

Scheme 2.7. Synthesis of biotinylated CEP derivative 2.25.

38 2.2.6. Syntheses of ethanolamine phospholipid CEP derivatives.

Phosphatidylethanolamines (PEs) are major components of certain membranes in brain

cells and in photoreceptor cells of the retina. Because levels of PEs are strictly regulated,

they are believed to have unique functional importance.22 In view of the reactivity of the

primary amino group of PEs and the abundance of DHA in brain and retina, we anticipate

that DHA-derived oxidatively truncated phospholipids containing reactive electrophilic

4-hydroxy-7-oxohept-5-enoates convert the primary amino group of PEs into CEP-PE

derivatives. We synthesized CEP-PEs to facilitate their detection and identification in

vivo. Reaction of DOHA-Fm with 1-palmityl-2-oleyl-sn-glycero-3-phosphoethanolamine

(POPE) or 1-palmityl-2-hydroxy-sn-glycero-3-phosphoethanolamine (lysoPE) followed

by deprotection of the intermediate Fm esters 2.26 or 2.28 with DBU (Scheme 2.8) delivered the CEP-PE 2.27 and lysoCEP-PE 2.29. Using these authentic standards, we

identified CEP-PEs in lipids extracted from retina. Details of those studies are reported in

Chapter 5.

O CO POPE 76% R O P O O Fm O 2.4 + or OH N lysoPE TEA, CHCl3 O C H 14 29 2.26, R = oleyl O 2.28, R = H

O CO R O P O OH 90% O OH N DBU, CHCl 3 O C H 14 29 2.27, R = oleyl O 2.29,R=H

Scheme 2.8. Syntheses of ethanolamine phospholipid CEP derivatives.

2.2.7. Synthesis of an active pentafluorophenyl ester of a lysyl CEP.

Pentafluorophenyl esters of protected amino acids are widely used in peptide synthesis.

We are interested in testing the ability of CEP modified peptides bound to major

39 histocompatibility proteins to elicit an immune response to antigen specific T-cells.

Furthermore, complexes of CEP modified peptides bound to constructs called “dimer X”,

that have major histocompatibility proteins fused to immunoglobin Fc constant regions,

can be used to fluorescently label antigen specific T-cells, and consequently enable their

quantitation by fluorescence activated cell sorting. The pentafluorophenyl ester 2.31 of a

CEP-Fm modified lysine was synthesized as a building block for construction of CEP

modified peptides. Reaction of DOHA-Fm with 6-amino-2-(9H-fluoren-9-

ylmethoxycarbonylamino)-hexanoic acid (Fmoc-Lys-OH) delivered 2.30. The latter was

then coupled with pentafluorophenol using the traditional DCC, DMAP method (Scheme

2.9).

FmO O H COOH H COOH F F Fmoc N Fmoc N O 75% O O 90% F N DOHA-Fm OFm C F OH, DCC 6 5 F F HN MeOH DMAP, CH Cl HOAC N 2 2 NH2 2.30 Fmoc 2.31 Scheme 2.9. Synthesis of a lysyl CEP pentafluorophenyl derivative.

2.2.8. Characterization of CEP-modified protein. To characterize the CEP adducts in

protein following modification with DOHAFm or DOHAFmSu and DBU deprotection,

the proteins were digested with trypsin (Protein:trypsin = 50:1, w/w) and analyzed by

liquid chromatography tandem MS (LC MS/MS) with a CapLC system (Micromass,

Beverly, MA) and a quadrupole time-of-flight mass spectrometer (QTOF2,

Micromass).23 Peptides were separated on a 75 μm x 5 cm Biobasic C18 column (New

Objective, Cambridge, MA) by using aqueous formic acid/ acetonitrile solvents, a flow

rate of 250 nL/min, and a gradient of 5-40% acetonitrile over 57 min followed by 80%

acetonitrile for 2 min. Protein identifications from MS/MS data was accomplished with

MASSLYNX 3.5 software (Waters), the Mascot search engine (Matrix Science) and the

40 Swiss-protein and National Center for Biotechnology information protein sequence

databases.24, 25 MS/MS analysis of modifications at specific m/z revealed peptide

sequences with unambiguous CEP or CEPH adducts on lysyl residues of the CEP-

proteins or CEPH-proteins. No CEPFm or CEPHFm modifications were found after

deprotection. We also analyzed CEPFm modified HSA before deprotection. The LC-MS

revealed 3 peptides with CEPFm modifications at m/z 725.281, 660.289 and 776.820 on

lysyl residues of the HSA peptides 25DAHKSEVAHR34, 234AFKAWAVAR242, and

247FPKAEFAEVSK257in CEPFmHSA (Appendix Figure S49). In addition, owing to spontaneous loss of the Fm group, 5 peptides with CEP modifications were found at m/z

636.248, 881.408, 571.262, 687.787 and 674.765 on lysyl residues of the HSA peptides

25DAHKSEVAHR34, 438KVPQVSTPTLVEVSR452, 234AFKAWAVAR242,

247FPKAEFAEVSK257, and 35FKDLGEENFK44 respectively in CEPFmHSA (Appendix

Figure S48).

2.2.9. 3 D protein structures of CEP modified HSA. LC-MS/MS revealed six CEP

modifications at m/z 589.266, 571.260, 674.762, 881.401, 687.784, 636.252 on lysyl

residues of the HSA peptides 161KYLYEIAR 168, 234AFKAWAVAR242,

35FKDLGEENFK44, 438KVPQVSTPTLVEVSR452, 247FPKAEFAEVSK257, and

25DAHKSEVAHR34 respectively. Notably, the sequence 234AFKAWAVAR242 is very

similar to the sequence 233ALKAWSVAR241 in BSA and identical with

234AFKAWAVAR242 in MSA. MS/MS analysis revealed modification on ALKAWSVAR

sequence of CEPH-BSA in preparations generated with different CEP ratios, implying

that this sequence is readily accessed and modified. Figure 2.2 represents a ball-and-stick

model of 3D visualization of HSA using PyMOL v0.99. X- ray crystallography of HSA

reveals a dimer structure with two identical units. We show all lysines as space-filling

41 structures in the left unit while only the lysines that become modified by CEP are shown

as space-filling structures in the right unit. The lysine residue in the 234AFKAWAVAR242

sequence is marked. The 3D structure shows that this lysine is located at the surface of

HSA and protrudes from the surface of the molecule. In addition, this sequence

incorporates nonpolar residues on both ends. The physical and chemical

characteristics of this specific sequence probably facilitate access by DOHA-Fm. Further

research is underway to synthesize this CEP modified peptide bound to major

histocompatibility complex (MHC) and to test its ability to elicit an immune response to

antigen specific T-cells.

K236

Figure 2.2. A ball-and-stick model of 3D structure for human serum albumin. All the lysines are shown as space-filling structures in the left unit. Only lysines that become

CEP modified are shown in the right unit. The lysine in the sequence which is common to

HSA, BSA and MSA is marked.

42 2.3. Conclusions.

The introduction of multiple CEP modifications of lysyl residues is readily achieved

through reaction of proteins with DOHA-Fm, a 9-fluorenylmethyl ester of 4,7- dioxoheptanoic acid, followed by deprotection of intermediate Fm esters of CEPs, without causing protein denaturation, by treatment with DBU. CEP-proteins and peptides, that are now readily available through the new methodology, are being evaluated as biomarkers for clinical prognosis of AMD, and in studies of their role in promoting AMD through the activation of B- and T-cells. The DOHA-Fm reagent is also useful for the preparation of CEPs linked to biotin as ligands to capture human scFv antibodies from phage display libraries, and to generate CEP modified ethanolamine phospholipids as standards to confirm their presence in retina. The characterization of CEP modified proteins was done by tandem mass spectrometry. LC-MS/MS analysis of modifications revealed peptide sequences with unambiguous CEP or CEPH adducts on lysyl residues of

HSA, BSA, MSA, CEO, myoglobin and GPDH. CEP peptides for HSA were described above, CEP and CEPH modified peptides from the remaining proteins are detailed in the experimental section.

43 2.4. Experimental Procedures

General Methods.

1H NMR spectra were recorded on a Varian Gemini spectrometer operating at 200, 300

MHz, or on a Varian Inova spectrometer operating at 400 MHz using CDCl3 (δ 7.24) or

CD3OD (δ 3.30) as internal standard. All chemical shifts are reported in parts per million

(ppm) on the δ scale relative to the internal standard used. 1H NMR spectral data are

tabulated in terms of multiplicity of proton absorption (s, singlet; d, doublet; dd, doublet

of doublets; dt, doublet of triplets; t, triplet; m, multiplet), coupling constants (Hz),

number of protons. 13C NMR spectra were recorded on a Varian Gemini spectrometer

operating at 75 MHz or on a Varian Inova spectrometer operating at 100 MHz using

CDCl3 (δ 77.0), or CD3OD (δ 49.0) as internal standard. High-resolution mass spectra

were recorded on a Kratos AEI MS25 RFA high-resolution mass spectrometer at 20 eV.

Chromatography was performed with ACS grade solvents (ethyl acetate, hexane,

chloroform, or methanol). Thin-layer chromatography (TLC) was performed on glass

plates precoated with silica gel (Kieselgel 60 F254, E. Merck, Darmstadt, West Germany).

Rf values are quoted for plates of thickness 0.25 mm. The plates were visualized by

viewing the developed plates under short-wavelength UV light, iodine, or a phospholipid

spray (Molybdenum spray).26 Flash column chromatography was performed on 230-400

mesh silica gel supplied by E. Merck. For all reactions performed in an inert atmosphere,

argon was used unless otherwise specified.

Bovine serum albumin (BSA, Cat. A2153), human serum albumin (HSA, Cat. A1887)

and chicken egg albumin (CEO, Cat. A5503) as well as myoglobin (Cat. M1882) and

glycerol-3-phosphate dehydrogenase (GPDH, Cat. G6880) were purchased from Sigma.

44 6-[1,3]Dioxolan-2-yl-4-oxo-hexanoic acid methyl ester (2.1). A solution of 2-(2-

bromoethyl)-1,3-dioxolane (1 g, 5.5 mmol) in anhydrous THF (2 mL) was added

dropwise to a flame dried 100 mL flask with Mg turnings (150 mg, 6.25 mmol) and THF

(4 mL) and a small piece of I2 were added under argon at room temperature to initiate the reaction. After adding a few drops, the reaction started as evidence by disappearance of the red-brown I2 color. Then THF (5 mL) was added to the flask. After completion of the

addition, more THF (15 mL) was added. The reaction mixture was stirred for another 1 h

and then cooled to -78 °C followed by slow addition of 3-carbomethoxypropionyl

chloride (710 mg, 4.7 mmol) dissolved in dry THF (2.5 mL). The resulting mixture was

stirred for another 40 min, then quenched with 30 mL of a saturated aqueous solution of

NH4Cl, and extracted with EtOAc (4 x 15 mL). The combined organic phase was washed

with brine, dried with MgSO4, and evaporated to obtain the crude product. The crude

product was purified by silica gel chromatography (30 % ethyl acetate in hexane, TLC: Rf

1 = 0.3) to give 714 mg (60 %) of pure 2.1. H NMR (CDCl3, 200 MHz) δ 4.85 (t, J = 4.3

Hz, 1H), 3.7-3.90 (4H), 3.62 (s, 3H), 2.67 (t, J = 7.48 Hz, 2H), 2.56 (t, J = 7.48 Hz, 4H),

13 1.93 (dt, J = 4.3, 7.48 Hz, 2H). C NMR (CDCl3, 50 MHz, APT) δ 207.81 (+) (CO),

173.09 (+) (COO), 103.06 (-) (CH), 64.83 (+) (CH2), 51.61 (-) (CH3), 36.87 (+) (CH2),

36.30 (+) (CH2), 27.62 (+) (CH2), 27.41 (+) (CH2). HRMS (FAB) (m/z) calcd for

+ C10H17O5 (MH ) 217.1076, found 217.1081.

6-[1,3]Dioxolan-2-yl-4-oxo-hexanoic acid (2.2). Ester 2.1 (390 mg, 1.8 mmol) in 10

mL of H2O/MeOH/THF (2:5:3, v/v/v) was stirred for 3 h with NaOH (367 mg, 9.2 mmol)

at room temperature. The reaction mixture was then acidified with 3 N HCl to pH 3.0 and

extracted with EtOAc (3 x 15 mL). The combined organic phase was washed with brine,

1 dried with MgSO4, and evaporated to give acid acetal 2.2 (360 mg, 90%). H NMR

45 (CDCl3, 200 MHz), δ 4.85 (t, J = 4.3 Hz, 1H), 3.7-3.90 (4H), 2.5-2.64 (6H), 1.93 (dt, J =

13 4.3, 7.48 Hz, 2H). C NMR (CDCl3, 100 MHz,) δ 207.81 (CO), 178.03 (COOH), 103.17

(CH), 64.97 (CH2), 36.76 (CH2), 36.36 (CH2), 27.71 (CH2), 27.51 (CH2). HRMS (FAB)

+ (m/z) calcd for C9H15O5 (MH ) 203.0919, found 203.0917.

6-[1,3]Dioxolan-2-yl-4-oxo-hexanoic acid 9H-fluoren-9-ylmethyl ester (2.3). (9H-

Fluoren-9-yl)-methanol (373 mg, 1.9 mmol) in 3 mL dry CH2Cl2 was slowly added to the

solution of dicyclohexylcarbodiimide (DCC, 295 mg, 1.425 mmol), dimethlamino

pyridine (DMAP, 58 mg, 0.475 mmol) and the acid 2.2 (96 mg, 0.475 mmol) in 5 mL dry

CH2Cl2. The resulting mixture was stirred for 72 h at room temperature. The solvent was

removed. Flash chromatography of the residue (30 % ethyl acetate in hexane, TLC: Rf =

1 0.3) gave the ester 2.3 (153 mg, 95 %). H NMR (CDCl3, 200 MHz) δ 7.77 (d, J = 6.74

Hz, 2H), 7.60 (d, J = 7.7 Hz, 2H), 7.42 (dd, J = 6.74, 7.7 Hz, 2H), 7.29 (dd, J = 6.74, 7.7

Hz, 2H), 4.90 (t, J = 4.3 Hz, 1H), 4.40 (d, J = 7.38 Hz, 2H), 4.21 (t, J = 7.38 Hz, 1H),

3.80-3.96 (4H), 2.62-2.73 (4H), 2.56 (t, J = 7.3 Hz, 2H), 1.98 (td, J = 7.3, 4.3 Hz, 2H).

13 C NMR (CDCl3, 50 MHz, APT), δ 207.76 (+) (CO), 172.62 (+) (COO), 143.69 (+) (C),

141.19 (+) (C), 127.69 (-) (CH), 127.04 (-) (CH), 125.00 (-) (CH), 119.92 (-) (CH),

103.12 (-) (CH), 66.45 (+) (CH2), 64.90 (+) (CH2), 46.67 (-) (CH), 36.89 (+) (CH2), 36.35

+ (+) (CH2), 27.90 (+) (CH2), 27.48 (+) (CH2). HRMS (FAB) (m/z) calcd for C23H24O5 (M )

+ 380.1624, found 380.1614; calcd for C23H25O5 (MH ) 381.1702, found 381.1711.

4,7-Dioxo-heptanoic acid 9H-fluoren-9-ylmethyl ester (DOHA-Fm, 2.4). Ester 2.3

(94 mg, 0.25 mmol) in 10 mL of AcOH/H2O (3:1, v/v) was stirred at 50 °C for 5 h. TLC

(5 % CHCl3 in MeOH): Rf = 0.5 showed the completion of the reaction.The solvent was

removed by rotary evaporation. Flash chromatography of the residue (25 % ethyl acetate

1 in hexane) gave 2.4 (73 mg, 88 %). H NMR (CDCl3, 400 MHz), δ 9.78 (s, 1H), 7.77 (d,

46 J = 6.74 Hz, 2H), 7.60 (d, J = 7.7 Hz, 2H), 7.42 (dd, J = 6.74, 7.7 Hz, 2H), 7.30 (dd, J =

6.74, 7.7 Hz, 2H), 4.39(d, J = 7.38 Hz, 2H), 4.20 (t, J = 7.38 Hz, 1H), 2.67-2.79 (8H). 13C

NMR (CDCl3, 100 MHz), δ 206.56 (CO), 200.27 (CHO), 172.53 (COO), 143.70 (C),

141.23 (C), 127.73 (CH), 127.07 (CH), 124.98 (CH), 119.96 (CH), 66.47 (CH2), 46.70

(CH), 37.42 (CH2), 36.87 (CH2), 34.53 (CH2), 27.93 (CH2). HRMS (FAB) (m/z) calcd for

+ C21H20O4 (M ) 336.1362, found 336.1361.

2-(2-Acetylamino-acetylamino)-6-[2-[2-(9H-fluoren-9-ylmethoxycarbonyl)-ethyl]- pyrrol-1-yl]-hexanoic acid methyl ester (2.5). Methyl 6-amino-2-((2- acetylamino)acetyl) amino) hexanoate (Ac-Gly-Lys-OMe, 25.6 mg, 0.08 mmol) in 1 mL methanol was added dropwise to DOHAFm (27 mg, 0.08 mmol) in 1.5 mL methanol.

The solution was stirred for 9 h at room temperature under argon. The solvent was removed by rotary evaporation. TLC (4 % methanol in chloroform): Rf = 0.34. The crude

compound was purified by silica gel chromatography (4 % methanol in chloroform ) to

1 give 35.8 mg (80%) of pure 2.5. H NMR (CDCl3, 400 MHz), δ 7.77 (d, J = 7.6 Hz, 2H),

7.60 (d, J = 7.6 Hz, 2H), 7.36-7.32 (dd, J = 7.6, 7.2 Hz, 2H), 7.27 (dd, J = 7.6, 7.2 Hz,

2H), 6.60 (dd, J = 2.4, 1.6 Hz, 1H), 6.54 (m, 1H), 6.28 (s, 1H), 6.09 (dd, J = 3.6, 2.4 Hz,

1H), 5.90 (m, 1H), 4.56-4.61 (m, 1H), 4.40 (d, J = 7.2 Hz, 2H), 4.21 (t, J = 7.2 Hz, 1H),

3.91 (dABq, J = 5.2, 14 Hz, 2H), 3.82 (t, J = 7.2 Hz, 2H), 3.76 (s, 3H), 2.65-2.86 (4H),

13 2.01 (s, 3H), 1.86 (2H), 1.66 (2H), 1.36 (2H). C NMR (CDCl3, 100 MHz), δ 172.98

(CO), 172.33 (CO), 170.58 (CO), 168.66 (CO), 143.74 (C), 141.31 (C), 130.73(C),

127.80 (CH), 127.12 (CH), 125.01 (CH), 120.37 (CH), 120.04 (CH), 106.99 (CH),

105.26 (CH), 66.47 (CH2), 52.53 (CH), 51.86 (CH3O), 46.78 (CH), 46.09 (CH2), 43.23

(CH2), 33.22 (CH2), 31.89 (CH2), 30.67 (CH2), 22.92 (CH2), 22.40 (CH2), 21.41 (CH3).

+ + HRMS (FAB) (m/z) calcd for C32H38N3O6 (MH ) 560.2755, found 560.2747.

47 2-(2-Acetylamino-acetylamino)-6-[2-(2-carboxy-ethyl)-pyrrol-1-yl]-hexanoic acid

methyl ester (CEP-dipep, 2.6). DBU (75 μL) was added to 2.5 (27 mg, 0.047 mmol) in

2.5 mL THF. The system was stirred for 6 h under argon. After the removal of solvent,

the crude compound was purified by silica gel chromatography (6 % methanol in

chloroform) to give 21 mg (86 %) of CEP-dipep. TLC (10 % methanol in chloroform): Rf

1 = 0.3; H NMR (CDCl3, 400 MHz), δ 7.18 (d, J = 8 Hz, 1H), 6.81 (m, 1H), 6.53 (m, 1H),

6.03 (dd, J = 3.2, 3.2 Hz, 1H), 5.87 (m, 1H), 4.53 (dt, J = 1H), 3.95 (dABq, J = 5.2, 13.6

Hz, 2H), 3.81 (t, J = 6.8 Hz, 2H), 3.70 (s, 3H), 2.65-2.86 (4H), 2.01 (s, 3H), 1.8-1.62

13 (4H), 1.36 (m, 2H). C NMR (CDCl3, 100 MHz), δ 176.01 (COOH), 172.40 (COO),

171.35 (CO), 169.28 (CO), 131.40 (C), 119.92 (CH), 107.25 (CH), 105.00 (CH), 52.50

(CH), 52.00 (CH3O), 45.66 (CH2), 43.19 (CH2), 32.98 (CH2), 31.44 (CH2), 30.90 (CH2),

+ + 22.94 (CH2), 22.07 (CH2), 21.37 (CH3). HRMS (FAB) (m/z) calcd for C18H28N3O6 (MH )

382.1978, found 382.1986.

Tryptic Digestion. CEP modified proteins were dissolved in NH4HCO3 buffer (8 M

urea/2M NH4HCO3) to make a final concentration 2-3 pmol/μL. Trypsin solution (0.1

μg/μL) was made by suspending sequencing grade modified porcine trypsin (20 μg)

(Promega, Cat. V511A) in trypsin resuspension buffer (Promega, Cat. V511A) (200 μL).

This trypsin solution was then added to protein solution in a ratio of 1:50 (w/w) enzyme/protein. After incubation at 37 °C for 24 h, the solution was centrifuged at 3000 rpm for 5 mins. The upper layer (10 μL) was subjected to LC-MS analysis.27

Carboxyethyl pyrrole human albumin adduct (CEP-HSA, 2.7). A solution of

DOHA-Fm (2 mg, 0.006 mmol) in DMF (750 μL) was added to HSA solution (0.08 mM,

1.5 mL) in PBS. The mixture was stirred under argon for 4 days. DBU (200 μL) was

48 added and the resulting mixture was stirred overnight under argon followed by two

successive 12 h dialyses (Mr cutoff 14000) against 500 mL 20 % DMF in 10 mM PBS

(pH 7.4) and two additional dialyses (12 h each) against 500 mL 10 mM PBS (pH 7.4) at

4 °C. The final protein concentration (1.80 mg/mL) was determined by the Pierce bicinchoninic acid (BCA) protein assay. The pyrrole concentration (187 μM) was determined by Ehrlich assay. LC-MS/MS revealed six CEP modifications at m/z 589.266,

571.260, 674.762, 881.401, 687.784, 636.252 on lysyl residues of the HSA peptides

161KYLYEIAR168, 234AFKAWAVAR242, 35FKDLGEENFK44, 438KVPQVSTPTLVEVS-

R452, 247FPKAEFAEVSK257, 25DAHKSEVAHR34 respectively (Appendix Figure S39).

No CEPFm modifications were found after deprotection. Figure 2.3 shows the tandem

MS spectrum of the doubly charged ion m/z 571.260 from a MS scan of tryptic digested

CEP-HSA. The spectrum shows a series of fragment ions (b ions and y ions) sufficient to

identify a CEP modification on Lys236 from HSA residues 234AFKAWAVAR242. This

particular sequence is very similar with the sequence 233ALKAWSVAR241 in BSA and is

exactly the same as in MSA. MS/MS analysis revealed unambiguous modification on the

ALKAWSVAR sequence of CEP-BSA and the AFKAWAVAR sequence of MSA.

Presumably, this sequence is readily accessed and modified.

49 ) Relative Abundance (%

Figure 2.3. Tandem MS characterization of the doubly charged ion m/z 571.260 from

a MS scan of tryptic digested CEP modified HSA shows series of fragment ions

sufficient to unambiguously identify a CEP modification on the lysyl residue (K236).

Asterisks denote fragment ions with the modified lysyl residue.

Carboxyethyl pyrrole mouse albumin adduct (CEP-MSA, 2.8). A solution of

DOHAFm (18.5 mg, 0.055 mmol) in DMF (8 mL)was added slowly to the solution of mouse serum albumin (100 mg) in 18 mL 10 mM PBS (pH 7.4). The mixture was stirred under argon for 4 days. DBU (360 μL) was added and the resulting mixture was stirred overnight under argon followed by two successive 24 h dialyses (Mr cutoff 14000) against 1 L 20 % DMF in 10 mM PBS (pH 7.4) and two additional dialyses (24 h each) against 1 L 10 mM PBS (pH 7.4) at 4 °C. The final protein concentration (2.43 mg/mL) was determined by the Pierce bicinchoninic acid (BCA) protein assay. The pyrrole concentration (210 μM) was determined by Ehrlich assay. LC-MS/MS revealed 15 CEP modifications at m/z 650.3296, 872.9611, 571.3492, 849.4302, 612.8407, 769.4069,

50 694.6888, 1047.0802, 857.4633 on lysyl residues of the MSA peptides 25EAHKSEI

AHR34, 206LDGVKEKALVSSVR219 (2 CEPs), 234AFKAWAVAR242, 243LSQTFPNADF

AEITKLATDLTK264, 258LATDLTKVNK267, 373LAKKYEATLEK383 (2 CEPs),

435YTQKAPQVSTPTLVEAAR452, 544EKQIKKQTALAELVK558 (3 CEPs), 550QTA

LAELVKHKPKATAEQLK 569 (3 CEPs) respectively (Appendix Figure S40). The sequence coverage was 35%. No CEPFm modifications were found after deprotection.

The tandem MS spectrum of AFKAWAVAR was shown in Figure 2.4. Relative Abundance (%)

Figure 2.4. Tandem MS characterization of the doubly charged ion m/z 571.3492 from

a MS scan of tryptic digested CEP modified MSA shows series of fragment ions

sufficient to unambiguously identify a CEP modification on the lysyl residue (K236).

Asterisks denote fragment ions with the modified lysyl residue.

Carboxyethyl pyrrole chicken egg albumin adduct (CEP-CEO, 2.9). A solution of

DOHAFm (18 mg, 0.054 mmol) in DMF (3.2 mL) was added slowly to the solution of

CEO (76 mg) in pH 7.4 PBS (10 mM, 7 mL). The mixture was stirred under argon for 4

days. DBU (140 μL) was added and the resulting mixture was stirred overnight under

51 argon followed by two successive 24 h dialyses (Mr cutoff 3500) against 1 L 20% DMF

in 10 mM pH 7.4 PBS and two more dialyses (24 h each) against 1 L 10 mM pH 7.4 PBS

at 4 °C. The final protein concentration (2.94 mg/mL) was determined by the Pierce

bicinchoninic acid (BCA) protein assay. The pyrrole concentration (93 μM) was determined by Ehrlich assay. LC-MS/MS revealed eight CEP modifications at m/z

540.2886, 801.7021, 839.3332, 808.0259, 610.2589, 630.8334, 459.1887 on lysyl residues of the CEO peptides 51TQINKVVR58, 85DILNQITKPNDVYSFSLASR104,

187AFKDEDTQAMPFR199, 200VTEQESKPVQMMYQIGLFR218, 219VASMASEKMK228,

277KIKVYLPR284, 285MKMEEK290 respectively (Appendix Figure S41). The sequence

coverage was 46%. No CEPFm modifications were found after deprotection.

Carboxyethyl pyrrole myoglobin adduct (CEP-myoglobin, 2.10). A solution of

DOHAFm (12 mg, 0.036 mmol) in DMF (3.6 mL) was added slowly to the solution of myoglobin (32.38 mg) in pH 7.4 PBS (10 mM, 5 mL). The mixture was stirred under argon for 4 days. DBU (140 μL) was added and the resulting mixture was stirred overnight under argon followed by two successive 24 h dialyses (Mr cutoff 3500) against

500 mL 20% DMF in 10 mM pH 7.4 PBS and two more dialyses (24 h each) against 500 mL 10 mM pH 7.4 PBS at 4 °C. The final protein concentration (0.65 mg/mL) was determined by Bio-Rad protein assay. The pyrrole concentration (91 μM) was determined by Ehrlich assay. LC-MS/MS revealed CEP modifications at m/z 671.2667, 604.9635,

815.2087, 557.4288, 785.6061, 899.6666, on lysyl residue of the myoglobin peptides

32LFTGHPETLEKFDKF KHLK50 (3 CEP modifications), 48HLKTEAEMK56, 63KHGT

VVLTALGGILK77, 79KGHHEAELKPLAQSHATK96, 119HPGDFGADAQGAMTKALE

LFR139, 140NDIAAKYKELGFQG153 (2 CEP modifications) (Appendix Figure S42). The

52 peptide sequence coverage achieved 91%. No CEPFm modifications were found after

deprotection.

Carboxyethyl pyrrole GPDH adduct (CEP-GPDH, 2.11). GPDH (30 mg) was added in 10 mL of 10 mM PBS (pH 7.4). After vortexing for 5 mins and centrifugation (4 °C) at 3000 rpm for 10 mins (GPDH does not dissolve well in PBS), 4 mL of clear solution was taken and DOHAFm (5.8 mg, 0.017 mmol) in DMF (4 mL) was added slowly to it.

The mixture was stirred under argon for 4 days. DBU (120 μL) was then added and the resulting mixture was stirred overnight under argon followed by two successive 24 h dialyses (Mr cutoff 3500) against 500 mL 20% DMF in 10 mM pH 7.4 PBS and two more dialyses (24 h each) against 500 mL 10 mM pH 7.4 PBS at 4 °C. The final protein concentration (0.29 mg/mL) was determined by the Bio-Rad protein assay. The pyrrole concentration (65 μM) was determined by Ehrlich assay. LC-MS/MS revealed six CEP modifications at m/z 655.3343, 669.3289, 923.9847, 639.3413, 740.8362 on lysyl residues of the GPDH peptides 105AGAHLKGGAKR115, 184TVDGPSGKLWR194,

198GAAQNIIPASTGAAKAVGK216, 258VVKQASEGPLK268, 321VVDLMVHMASKE332

respectively (Appendix Figure S43). The peptide sequence coverage achieved 45%. No

CEPFm modifications were found after deprotection.

6-[2-[2-(9H-fluoren-9-ylmethoxycarbonyl)-ethyl]-pyrrol-1-yl]-hexanoic acid (2.12).

6-Aminocaproic acid (10.8 mg, 0.082 mmol) in water (400 μL) was slowly added to

DOHA-Fm (21 mg, 0.0625 mmol) in methanol (600 μL). The solution became cloudy

with the addition. The heterogeneous system was stirred for 48 h under argon at room

temperature during which became homogenous. The solution was extracted with CH2Cl2,

washed with brine, dried with Na2SO4 and evaporated. The yellowish residue was loaded

onto silica gel in a sintered-glass funnel with chloroform and washed with chloroform (15

53 mL), 10 % ethyl acetate in hexane (15 mL), 20 % ethyl acetate in hexane (15 mL) and 50

% ethyl acetate in hexane (60 mL) successively. The fractions eluted with 50 % ethyl

acetate/hexane were collected and dried to give 21.8 mg (81 %) acid 2.12. TLC (ethyl

1 acetate/hexane, 2:3 v/v): Rf = 0.22. H NMR (CDCl3, 400 MHz), δ 7.77 (d, J = 6.74 Hz,

2H), 7.58 (d, J = 7.7 Hz, 2H), 7.42 (dd, J = 7.7, 7.2 Hz, 2H), 7.30 (dd, J = 6.74, 7.2 Hz,

2H), 6.58 (dd, J = 3.2, 3.6 Hz, 1H), 6.06 (dd, J = 3.2, 3.2 Hz, 1H), 5.87 (m, 1H), 4.42 (d,

J = 7.2 Hz, 2H), 4.22 (t, J = 7.2 Hz, 1H), 3.79 (t, J = 7.2 Hz, 2H), 2.72-2.84 (4H). 2.34 (t,

13 J = 7.2 Hz, 2H), 1.75-1.34 (6H). C NMR (CDCl3, 100 MHz), δ 177.40 (COOH), 172.90

(COO), 143.73 (C), 141.20 (C), 130.66 (C), 127.78 (CH), 127.10 (CH), 125.00 (CH),

120.34 (CH), 120.04 (CH), 106.88 (CH), 105.20 (CH), 66.40 (CH2), 46.79 (CH2), 46.28

(CH), 33.45 (CH2), 33.20 (CH2), 31.01 (CH2), 26.23 (CH2), 24.26 (CH2), 21.44 (CH2).

+ HRMS (FAB) (m/z) calcd for C27H30NO4 (MH ) 432.2175, found 432.2190.

6-[2-[2-(9H-fluoren-9-ylmethoxycarbonyl)-ethyl]-pyrrol-1-yl]-hexanoic acid 2,5-

dioxo-pyrrolidin-1-yl ester (CEPFmSu, 2.13). Acid 2.12 (15 mg, 0.035 mmol) and N-

hydroxysuccinimide (4.5 mg, 0.039 mmol), DCC (7.5 mg, 0.036 mmol) were dissolved in

dry CH2Cl2 (7.5 mL) under Argon. The clear solution became cloudy after 15 minutes.

The reaction mixture was stirred for 3.5 h. Solvent was then removed by evaporation.

The crude product was purified by silica gel chromatography with ethyl acetate/hexane

(2:3, v/v) to deliver 16.6 mg (90 %) active ester 2.13. TLC (ethyl acetate/hexane, 2:3): Rf

1 = 0.25. H NMR (CDCl3, 400 MHz), δ 7.77 (d, J = 6.74 Hz, 2H), 7.58 (d, J = 7.7 Hz, 2H),

7.42 (dd, J = 7.7, 7.2 Hz, 2H), 7.30 (dd, J = 6.74, 7.2 Hz, 2H), 6.58 (dd, J = 3.2, 3.6 Hz,

1H), 6.06 (dd, J = 3.2, 3.2 Hz, 1H), 5.87 (m, 1H), 4.42 (d, J = 7.2 Hz, 2H), 4.22 (t, J = 7.2

Hz, 1H), 3.79 (t, J = 7.2 Hz, 2H), 2.72-2.86 (8H), 2.60 (t, J = 7.2 Hz, 2H), 1.81-1.70 (4H),

13 1.48-1.40 (m, 2H). C NMR (CDCl3 100 MHz), δ 172.79 (COO), 169.10 (COO), 168.39

54 (CO), 143.75 (C), 141.29 (C), 130.69 (C), 127.77 (CH), 127.10 (CH), 125.01 (CH),

120.34 (CH), 120.01 (CH), 106.92 (CH), 105.24 (CH), 66.36 (CH2), 46.79 (CH2), 46.21

(CH), 33.31 (CH2), 30.86 (CH2), 30.72 (CH2), 25.87 (CH2), 25.55 (CH2), 24.25 (CH2),

+ 21.44 (CH2). HRMS (FAB) (m/z) calcd for C31H33N2O6 (MH ) 529.2338, found 529.2340.

6-(2-carboxyethyl-1-pyrrolyl)-hexanoyl BSA amide (CEPH-BSA, 2.14) A solution of CEPFmSu (1.4 mg) in DMF (150 μL) was added to BSA (3 mg/mL, 1 mL) in PBS (10 mM, pH 7.4) solution. The cloudy solution became homogenous with overnight stirring.

After 2 days, DBU (25 μL) was added and the resulting mixture was stirred overnight under argon followed by two successive 12 h dialyses (Mr cutoff 14000) against 500 mL

20 % DMF in PBS (10 mM, pH 7.4) and two additional dialyses (12 h each) against 500 mL PBS (10 mM, pH 7.4) at 4 °C. The final protein concentration (1.34 mg/mL) was determined by modified Lowry protein assay. The pyrrole concentration (134 μM) was determined by Ehrlich assay. LC-MS/MS revealed nine CEPH modifications at m/z

594.9934, 618.8188, 541.8060, 937.9577, 526.7835, 612.3604, 689.4319, 765.3984,

964.5088 corresponding to the BSA peptides 400LKHLVDEPQNLIK412,

233ALKAWSVAR 241, 242LSQKFPK248, 437KVPQVSTPTLVEVSR451, 452SLGKVGTR459,

490TPVSEKVTK498, 548KQTALVELLK557, 246FPKAEFVEVTK256, 249AEFVEVTKLV

TDLTK263 respectively and unambiguously confirmed CEPH adducts on lysyl residues

(Appendix Figure S44). No CEPHFm modifications were found after deprotection. The tandem MS spectrum of ALKAWSVAR is shown in Figure 2.5.

55 Relative Abundance (%)

Figure 2.5. Tandem MS characterization of the doubly charged ion m/z 618.8188 from a

MS scan of tryptic digested CEPH-BSA shows series of fragment ions sufficient to

unambiguously identify a CEP modification on lysyl residue (K235). Asterisks denote

fragment ions with the modified lysyl residue.

Preparation of carboxyethyl pyrrole hexanoic acid 2,5-dioxo-pyrrolidin-1-yl

mouse serum albumin (CEPH-MSA, 2.15). A solution of CEPFmSu (630 μg) in DMF

(150 μL) was added in MSA (2 mg/mL, 1 mL) in PBS (10 mM, pH 7.4). The cloudy

solution became clear with overnight stirring. After 2 days, DBU (25 μL) was added, and

the resulting mixture was stirred overnight under argon followed by two successive 12 h

dialyses (Mr cutoff 3500) against 500 mL 20% DMF in PBS (10 mM, pH 7.4) and two

more dialyses (12 h each) against 500 mL PBS (10 mM, pH 7.4) at 4 °C. The final

protein concentration (2.06 mg/mL) was determined by modified Lowry protein assay.

The pyrrole concentration (69.5 μM) was determined by Ehrlich assay. LC-MS/MS

revealed four CEPH modifications at m/z 512.2829, 627.8604, 608.8177, 732.3707 on

56 lysyl residues of the MSA peptides 206LDGVKEK 212, 234AFKAWAVAR242, 376KYEATL

EK383, 435YTQKAPQVSTPTLVEAAR452, respectively (Appendix Figure S45). No

CEPHFm modifications were found after deprotection.

Preparation of carboxyethyl pyrrole hexanoic acid 2,5-dioxo-pyrrolidin-1-yl

chicken egg albumin (CEPH-CEO, 2.16). A solution of CEPFmSu (1.0 mg) in DMF

(150 μL) was added in CEO (4 mg/mL, 1 mL) in PBS (10 mM, pH 7.4). After the

coupling underwent for 2 days, the solution was still not clear until the addition of DBU

(25 μL). The system was stirred overnight under argon followed by two successive 12 h

dialyses (Mr cutoff 14000) against 500 mL 20% DMF in PBS (10 mM, pH 7.4) and two

more dialyses (12 h each) against 500 mL PBS (10 mM, pH 7.4) at 4 °C. The final

protein concentration (3.46 mg/mL) was determined by modified Lowry protein assay.

The pyrrole concentration (85.4 μM) was determined by Ehrlich assay. LC-MS/MS revealed a CEPH modification at m/z 596.8715 on a lysyl residue of the CEO peptide

51TQINKVVR 58 (Appendix Figure S46). No CEPHFm modifications were found after

deprotection.

Preparation of carboxyethyl pyrrole hexanoic acid 2,5-dioxo-pyrrolidin-1-yl

GPDH adduct (CEPH-GPDH, 2.17). GPDH (10 mg) was added in PBS (10 mM, 10

mL). After vortexing for 5 min and centrifugation (4 °C) at 3000 rpm for 10 mins, the

upper clear solution (2 ml) was separated and CEPFmSu (770 μg) in DMF (300 μL) was

added. The cloudy solution became clear after 2 days. Then DBU (25 μL) was added and

the resulting mixture was stirred overnight under argon followed by two successive 12 h

dialyses (Mr cutoff 3500) against 500 mL 20% DMF in PBS (10 mM, pH 7.4) and two more dialyses (12 h each) against 500 mL PBS (10 mM, pH 7.4) at 4 °C. The final protein concentration (0.32 mg/mL) was determined by modified Lowry protein assay.

57 The pyrrole concentration (32.5 μM) was determined by Ehrlich assay. LC-MS/MS revealed CEPH modifications at m/z 635.0367, 712.5500, 919.7650, 572.9763, 899.6923,

726.0422, 980.7797, 731.1005, 941.7422, 579.9560, 508.9000, 696.1018, 797.5823 on lysyl residues of the GPDH peptides 1VKVGVNGFGR10, 53FHGTVKAENGK63,

64LVINGKAITIFQER77, 105AGAHLKGGAK114, 160VIHDHFGIVEGLMTTVHAITA

TQKTVDGPSGK191, 184TVDGPSGKLWR194, 198GAAQNIIPASTGAAKAVGK216,

213AVGKVIPELNGK224, 217VIPELNGKLTGMAFR231, 249AAKYDDIK256,

252YDDIKK257, 258VVKQASEGPLK268, and 321VVDLMVHMASKE332 (Appendix

Figure S47). No CEPHFm modifications were found after deprotection. The peptide

sequence coverage achieved 68%.

[4-[5-(2-oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-butyl]-

carbamic acid tert-butyl ester (2.18). N-Boc-1,4-butanediamine (20 mg, 0.106 mmol)

in CH2Cl2 (2 mL) was added in d-biotin p-nitrophenyl ester (38 mg, 0.10 mmol) in

CH2Cl2 (2 mL). The mixture was stirred for 10 h. TLC analysis (30 % ethyl acetate in hexane) showed the completion of the reaction with a new spot at Rf = 0.25 generated.

After removal of the solvent by rotary evaporation, the crude compound was purified by flash chromatography (30 % ethyl acetate in hexane) to give 28 mg (80 %) 2.18. 1H

NMR (CD3OD, 400 MHz), δ 4.47-4.50 (m, 1H), 4.28-4.31 (m, 1H), 3.2 (m, 1H), 3.16 (t,

J = 7.2 Hz, 2H), 3.03 (t, J = 6.8 Hz, 2H), 2.92 (dd, J = 12.4, 4.8 Hz, 1H), 2.68-2.71 (d, J

= 12.8 Hz, 1H), 2.19 (t, J = 7.6 Hz, 2H), 1.44-1.7 (10H), 1.42 (s, 9H).

5-(2-Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoic acid (4-amino-butyl)- amide (2.19). Trifluoroacetic acid (100 μL) was added to 2.18 (6 mg) in CH2Cl2 (400

μL). After stirring for 3 h, TLC (20% methanol in chloroform, Rf = 0.1) showed the

reaction was completed. The solvent was removed by rotary evaporation. The yellowish

58 residue was basified with 1N NaOH to pH 8.5. After removal of the solvent, the residue

was triturated with methanol and the methanol extract was concentrated and dried to give

1 4 mg (90%) 2.19. H NMR (CD3OD, 400 MHz), δ 4.46-4.51 (m, 1H), 4.28-4.31 (m, 1H),

3.2 (m, 1H), 3.16 (t, J = 7.2 Hz, 2H), 2.9 (dd, J = 12.4, 4.8 Hz, 1H), 2.68-2.71 (d, J = 12.4

Hz, 1H), 2.65 (t, J = 7.2 Hz, 2H), 2.19 (t, J = 7.6 Hz, 2H), 1.34-1.7 (10H).

3-(1-[4-[5-(2-Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-butyl]-

1H-pyrrol-2-yl)-propionic acid 9H-fluoren-9-yl-methyl ester (2.20). To amine 2.19 (4 mg, 0.012 mmol) in methanol (150 μL), was added DOHA-Fm (4 mg, 0.0119 mmol) in methanol (150 μL). The solution was stirred for 9 h at room temperature under argon.

The solvent was then removed by rotary evaporation. The crude compound was purified by silica gel chromatography (5 % methanol in chloroform, TLC: Rf = 0.2) to give 6 mg

1 (84 %) of pure 2.20. H NMR (CDCl3, 400 MHz), δ 7.70 (d, J = 7.6 Hz, 2H), 7.50 (dd, J

= 7.6, 1.2 Hz, 2H), 7.34 (dd, J = 7.6, 7.2 Hz, 2H), 7.27-7.23 (ddd, J = 7.6, 7.2, 1.2 Hz,

2H), 6.51 (dd, J = 1.6, 2.8 Hz, 1H), 5.98 (dd, J = 2.8, 3.6 Hz, 1H), 5.79 (m, 1H), 5.74 (m,

1H), 5.58 (s, 1H), 4.82 (s, 1H), 4.40 (m, 1H), 4.36 (d, J = 7.2 Hz, 2H), 4.20 (m, 1H), 4.15

(t, J = 7.2 Hz, 1H), 3.76 (t, J = 7.2 Hz, 2H), 3.15 (m, 2H), 3.05 (m, 1H), 2.84-2.79 (dd, J

= 4.8, 12.8 Hz, 1H), 2.78-2.76 (m, 2H), 2.71-2.66 (m, 2H), 2.64-2.61 (d, J = 12.8 Hz, 1H),

2.06-2.10 (dt, J = 3.6, 7.2 Hz, 2H), 1.72-1.36 (10H). HRMS (FAB) (m/z) calcd for

+ + C35H43N4O4S (MH ) 615.3005, found 615.2995.

3-(1-[4-[5-(2-Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-butyl]-

1H-pyrrol-2-yl)-propionic acid (2.21). DBU (5 μL) was added to 2.20 (6 mg, 0.01

mmol) in THF (500 μL) and the resulting mixture was stirred for 2 h. TLC (10 % methanol in chloroform, Rf = 0.3) showed the completion of the reaction by

59 disappearance of the UV active starting spot. The corresponding acid was purified by

flash chromatography (7 % methanol in chloroform) to provide 3.7 mg (85 %) acid 2.21.

1 H NMR (CD3OD, 400 MHz), δ 6.57 (dd, J = 2.8, 1.6 Hz, 1H), 5.92 (dd, J = 3.2, 2.8 Hz,

1H), 5.79 (m, 1H), 4.60 (3H), 4.48 (m, 1H), 4.27 (m, 1H), 3.86 (t, J = 7.2 Hz, 2H), 3.19-

3.15 (3H), 2.94-2.89 (dd, J = 4.8, 12.8 Hz, 1H), 2.86-2.82 (m, 2H), 2.71-2.67 (d, J = 12.8

Hz, 1H), 2.60-2.56 (m, 2H), 2.20-2.17 (t, J = 7.2 Hz, 2H), 1.72-1.36 (10H). HRMS (FAB)

+ + + (m/z) calcd for C21H33N4O4S (MH ) 437.2222, found 437.2193; calcd for C21H31N4O3S

(M+-OH) 419.2117, found 419.2102.

4,7-Dioxo-heptanoic acid methyl ester (DOHA-Me, 2.22). Ester 2.1 (22.5 mg, 0.104 mmol) in 5 mL of AcOH/H2O (3:1, v/v) was stirred at 50 °C for 5 h. TLC (3% MeOH in

CHCl3, Rf = 0.4) showed the completion of the reaction. The solvent was removed by

rotary evaporation. Flash chromatography of the residue (25 % ethyl acetate in hexane)

1 gave 2.22 (15.6 mg, 87 %). H NMR (CDCl3, 200 MHz), δ 9.78 (s, 1H), 3.67 (s, 3H),

+ + 2.56-2.81 (8H); HRMS (FAB) (m/z) calcd for C8H11O4 (M -H) 171.0658, found

+ + 171.0656; calcd for C8H13O5 (M + OH) 189.0763, found 189.0783.

5-(2-Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoic acid (6-amino-hexyl)- amide (2.23). 1,6-Diaminohexane (270 mg, 2.324 mmol) in 10 mL pyridine-H2O (9:1, v/v) was added slowly to d-biotin p-nitrophenyl ester (Sigma) (100 mg, 0.274 mmol) in

28 20 mL pyridine-H2O (9:1,v/v). The clear yellow solution was stirred for 24 h at room

temperature. The solvent was removed by rotary evaporation. TLC (methanol (saturated

with NH3): CHCl3, 3:7, v/v), Rf = 0.24. The crude product was purified by silica gel

chromatography (30 % methanol saturated with NH3 in chloroform) to give 84 mg (90 %)

1 of pure 2.23. H NMR (CD3OD, 400 MHz), δ 4.46-4.51 (m, 1H), 4.28-4.31 (m, 1H),

3.20 (m, 1H), 3.16 (t, J = 7.2 Hz, 2H), 2.9 (dd, J = 12.4, 4.8 Hz, 1H), 2.68-2.71 (d, J =

60 12.4 Hz, 1H), 2.65 (t, J = 7.2 Hz, 2H), 2.19 (t, J = 7.6 Hz, 2H), 1.34-1.7 (14H). 13C NMR

(CD3OD, 100 MHz), δ 175.97 (CO), 166.10 (CO), 63.36 (CH), 61.60 (CH), 57.01 (CH),

42.3 (CH2), 42.0 (CH2), 40.2 (CH2), 36.8 (CH2), 33.2 (CH2), 30.50 (CH2), 29.8 (CH2),

29.5 (CH2), 28.8 (CH2), 28.6 (CH2), 28.0 (CH2). HRMS (FAB) (m/z) calcd for

+ + C16H31N4O2S (MH ) 343.2168, found 343.2165.

3-(1-[6-[5-(2-Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-hexyl]-

1H-pyrrol-2-yl)-propionic acid methyl ester (2.24). Amine 2.23 (45 mg, 0.13 mmol) in

1 mL methanol was added to DOHA-Me (23 mg, 0.13 mmol) in 1.5 mL methanol. The

solution was stirred for 9 h at room temperature under argon. The solvent was removed

by rotary evaporation. The crude product was purified by silica gel chromatography (5 %

1 methanol in chloroform, TLC: Rf = 0.18) to give 47 mg (75 %) of pure 2.24. H NMR

(CD3OD, 400 MHz), δ 6.52 (dd, J = 2.0, 2.8 Hz, 1H), 5.9 (dd, J = 3.2, 2.8 Hz, 1H), 5.75

(m, 1H), 4.46-4.49 (dd, J = 8.0, 4.0 Hz, 1H), 4.29 (dd, J = 8.0, 4.8 Hz, 1H), 4.28-4.3 (m,

1H), 3.82 (t, J = 7.6 Hz, 2H), 3.65 (s, 3H), 3.1-3.2 (3H), 2.9 (dd, J = 12.4, 4.8 Hz, 1H),

2.85 (t, J = 7.2 Hz, 2H), 2.68-2.71 (d, J = 12.4 Hz, 1H), 2.65 (t, J = 7.2 Hz, 2H), 2.19 (t, J

= 7.6 Hz, 2H), 1.34-1.7 (14H).

3-(1-[4-[5-(2-Oxo-hexahydro-thieno[3,4-d]imidazol-4-yl)-pentanoylamino]-hexyl]-

1H-pyrrol-2-yl)-propionic acid (2.25). Sodium hydroxide (16 mg, 0.4 mmol) was added to 2.24 (47 mg, 0.1 mmol) in 1 mL absolute ethanol and the resulting mixture was stirred for 4 h at room temperature. TLC (10 % methanol in chloroform, Rf = 0.3) showed the

completion of the reaction by disappearance of the starting spot. After removal of the

solvent, the residue was acidified with 3 N HCl to pH = 3 and then extracted with ethyl

acetate. The ethyl acetate extract was washed with brine, dried with MgSO4, and

1 concentrated to afford 38 mg (84 %) 2.25. H NMR (CD3OD, 400 MHz), δ 6.57 (dd, J =

61 2.0, 2.4 Hz, 1H), 5.93 (dd, J = 2.4, 3.6 Hz, 1H), 5.80 (m, 1H), 4.46 (dd, J = 7.6, 5.2 Hz,

1H), 4.29 (dd, J = 4.8, 7.6 Hz, 1H), 3.84 (t, J = 7.6 Hz, 2H), 3.1-3.2 (3H), 2.9 (dd, J =

12.4, 4.8 Hz, 1H), 2.85 (t, J = 7.2 Hz, 2H), 2.68-2.71 (d, J = 12.4 Hz, 1H), 2.61 (t, J = 7.2

13 Hz, 2H), 2.19 (t, J = 7.2 Hz, 2H), 1.34-1.7 (14H). C NMR (CD3OD, 100 MHz),

δ 176.82 (CO), 175.97 (CO), 166.10 (CO), 132.00 (C), 121.22 (CH), 107.52 (CH),

106.08 (CH), 63.36 (CH), 61.60 (CH), 57.01 (CH), 47.19 (CH2), 41.03 (CH2), 40.22

(CH2), 36.82 (CH2), 34.48 (CH2), 32.54 (CH2), 30.30 (CH2), 29.78 (CH2), 29.51 (CH2),

27.62 (CH2), 27.44 (CH2), 26.95 (CH2), 22.55 (CH2). HRMS (FAB) (m/z) calcd for

+ + C23H37N4O4S (MH ) 465.2530, found 465.2524.

Octadec-9-enoic acid 2-[(2-[2-[2-(9H-fluoren-9-ylmethoxycarbonyl)-ethyl]-pyrrol-

1-yl]-ethoxy)-hydroxy-phosphoryloxy]-1-hexadecanoyloxymethyl-ethyl ester (2.26).

Triethylamine (TEA) (25 μL, 0.247 mmol) was added to POPE (50 mg, 0.065 mmol) in

500 μL CHCl3, then DOHAFm (22 mg, 0.065 mmol) in 500 μL CHCl3 was added. The

resulting mixture was stirred 20 h under Argon. After evaporation of solvent, the crude

product was purified by silica gel chromatography (10 % methanol in chloroform, TLC:

1 Rf = 0.2) to give 50 mg (76 %) of pure 2.26. H NMR (CD3OD:CDCl3 = 1:1, 400 MHz),

δ 7.74 (d, J = 7.6 Hz, 2H), 7.53 (d, J = 7.6 Hz, 2H), 7.36 (dd, J = 7.6, 7.6 Hz, 2H), 7.27

(dd, J = 7.6, 7.6 Hz, 2H), 6.63 (dd, J = 2.8, 1.6 Hz, 1H), 5.94 (dd, J = 3.2, 2.8 Hz, 1H),

5.77 (m, 1H), 5.28 (m, 2H), 4.36 (d, J = 6.8 Hz, 2H), 4.28 (m, 1H), 4.17 (t, J = 6.8 Hz,

1H), 4.05-3.98 (6H), 3.68 (t, J = 6.4 Hz, 2H), 2.86-2.82 (t, J = 7.2 Hz, 2H), 2.73-2.69 (t, J

= 7.2 Hz, 2H), 2.25-2.21 (4H), 2.0-1.94 (4H), 1.57 (4H), 1.21 (44H), 0.84 (t, J = 6.4 Hz,

13 6H). C NMR (CD3OD:CDCl3 = 1:1, 50 MHz, APT), δ 175.21 (+) (CO), 174.84 (+)

(CO), 174.81 (+) (CO), 145.08 (+) (C), 142.68 (+) (C), 132.30 (+) (C), 131.24 (-) (CH),

130.95 (-) (CH), 129.13 (-) (CH), 128.53 (-) (CH), 126.31 (-) (CH), 121.30 (-) (CH),

62 121.22 (-) (CH), 108.49 (-) (CH), 106.77 (-) (CH), 71.86 (-) (CH), 67.96 (+) (CH2), 66.62

(+) (CH2), 64.60 (+) (CH2), 64.05 (+) (CH2), 48.10 (+) (CH2), 47.82 (-) (CH), 35.47 (+)

(CH2), 35.34 (+) (CH2), 34.66 (+) (CH2), 33.27 (+) (CH2), 31.03 (+) (CH2), 30.85 (+)

(CH2), 30.70 (+) (CH2), 30.66 (+) (CH2), 30.61 (+) (CH2), 30.49 (+) (CH2), 28.49 (+)

(CH2), 26.24 (+) (CH2), 26.20 (+) (CH2), 23.98 (+) (CH2), 22.64 (+) (CH2), 15.14 (-)

+ + (CH3). HRMS (FAB) (m/z) calcd for C60H91NNa2O10P [(M-H)Na2 ] 1062.6177, found

1062.6.

Octadec-9-enoic acid 2-([2-[2-(2-carboxy-ethyl]-pyrrol-1-yl]-ethoxy)-hydroxy-

phosphoryloxy]-1-hexadecanoyloxymethyl-ethyl ester (CEP-PE, 2.27). DBU (20 μL,

0.13 mmol) was added to 2.26 (24 mg, 0.025 mmol) in 500 μL CHCl3. After 3 h, the

reaction was completed and the system was diluted by 1.5 mL CHCl3. The solution was

washed with pH 5.5 phosphate buffer (2 mL). The organic phase was washed with brine,

dried over sulfate, then vacuum-filtered, concentrated, and purified by flash

chromatography (15 % methanol in chloroform, TLC: Rf = 0.18) to yield 19 mg (90 %) of

1 pure 2.27. H NMR (CD3OD:CDCl3 = 1:1, 400 MHz), δ 6.56 (m, 1H), 5.96 (m, 1H),

5.83 (m, 1H), 5.30 (m, 2H), 5.15 (m, 1H), 4.33 (m, 1H), 4.05-3.98 (6H), 3.81 (m, 2H),

2.83 (m, 2H), 2.58 (m, 2H), 2.27 (t, J = 7.2 Hz, 4H), 2.0-1.94 (4H), 1.57 (4H), 1.21 (44H),

13 0.84 (t, J = 6.4 Hz, 6H). C NMR (CD3OD:CDCl3 = 1:1, 100 MHz), δ 180.36 (COOH),

174.49 (CO), 174.08 (CO), 133.54 (C), 130.46 (CH), 130.14 (CH), 120.98 (CH), 107.26

(CH), 105.05 (CH), 71.00 (CH), 65.84 (CH2), 64.03 (CH2), 63.11 (CH2), 47.29 (CH2),

34.70 (CH2), 34.56 (CH2), 32.40 (CH2), 32.42 (CH2), 30.23 (CH2), 30.17 (CH2), 30.00

(CH2), 29.80 (CH2), 29.63 (CH2), 27.66 (CH2), 25.40 (CH2), 23.13 (CH2), 14.31 (CH3).

+ + HRMS (FAB) (m/z) calcd for C46H82NNaO10P (MNa ) 862.5574, found 862.5550; calcd

+ + for C46H81NNa2O10P [(M-H)Na2 ] 884.5394, found 884.5291.

63 Hexadecanoic acid 3-[(2-[2-[2-(9H-fluoren-9-ylmethoxycarbonyl)-ethyl]-pyrrol-1-

yl]-ethoxy)-hydroxy-phosphoryloxy]-2-hydroxy-propyl ester (2.28). Triethylamine

(TEA) (12 μL, 0.116 mmol) was added to lyso-PE (42 mg, 0.093 mmol) in 500 μL

CHCl3, then DOHAFm (26 mg, 0.077 mmol) in 500 μL CHCl3 was added to the mixture.

The cloudy system was stirred 20 h under Ar. After evaporation of solvent, the crude

product was purified by silica gel chromatography (10 % methanol in chloroform, TLC:

1 Rf = 0.2) to give 40 mg (69 %) of pure 2.28. H NMR (CDCl3, 400 MHz), δ 7.70 (d, J =

7.6 Hz, 2H), 7.52 (d, J = 7.6 Hz, 2H), 7.35 (dd, J = 7.6, 7.6 Hz, 2H), 7.25 (dd, J = 7.6, 7.6

Hz, 2H), 6.58 (m, 1H), 5.94 (m, 1H), 5.74 (m, 1H), 5.28 (m, 2H), 4.31 (d, J = 6.8 Hz, 2H),

4.14 (t, J = 6.8 Hz, 1H), 4.03-3.60 (9H), 2.82-2.78 (t, J = 7.2 Hz, 2H), 2.69-2.65 (t, J =

7.2 Hz, 2H), 2.21-2.17 (t, J = 7.2 Hz, 4H), 1.54-1.45 (m, 2H), 1.23-1.19 (24H), 0.86 (t, J

13 = 6.8 Hz, 3H). C NMR (CDCl3, 100 MHz), δ 173.69 (CO), 173.42 (CO), 143.69 (C),

141.22 (C), 131.07 (CH), 127.77 (CH), 127.09 (CH), 125.02 (CH), 120.97 (CH), 119.98

(CH), 107.20 (CH), 105.34 (CH), 77.20 (CH) 69.07 (CH2), 66.66 (CH2), 65.37 (CH2),

64.30 (CH2), 46.65 (CH2), 46.35 (CH), 33.97 (CH2), 33.11 (CH2), 31.92 (CH2), 29.73

(CH2), 29.67 (CH2), 29.58 (CH2), 29.37 (CH2), 29.23 (CH2), 24.83 (CH2), 22.69 (CH2),

+ + 21.23 (CH2), 14.11 (CH3). HRMS (FAB) (m/z) calcd for C41H58NNaO9P (MNa )

+ + 776.3904, found 776.3939; calc. for C41H57NNa2O9P (MNa2 ) 798.3724, found

798.3717.

Hexadecanoic acid 3-([2-[2-(2-carboxy-ethyl)-pyrrol-1-yl]-ethoxy]-hydroxy-

phosphoryloxy)-2-hydroxy-propyl ester (lysoCEP-PE, 2.29). DBU (20 μL, 0.13 mmol) was added to 2.28 (24 mg, 0.03 mmol) in 500 μL CHCl3. After 3 h, the reaction was

completed and the mixture was diluted by 1.5 mL CHCl3. The solution was washed with

64 pH 5.5 phosphate buffer (2 mL). The organic layer was washed with brine, dried over

magnesium sulfate and vacuum-filtered, concentrated, and purified by flash

chromatography (CHCl3:MeOH:H2O = 65:25:4, v/v, TLC: Rf = 0.18) to yield 16 mg (95

1 %) of pure lysoCEP-PE. H NMR (CD3OD:CDCl3 = 1:1, 400 MHz), δ 6.60 (m, 1H), 5.96

(m, 1H), 5.83 (m, 1H), 4.10-4.0 (5H), 3.74-3.6 (m, 2H), 3.6-3.5 (m, 2H), 2.8-2.9 (m, 2H),

2.68-2.65 (m, 2H), 2.32 (t, J = 6.8 Hz, 2H), 1.57 (m, 2H), 1.23 (24H), 0.85 (t, J = 7.2 Hz,

13 3H). C NMR (CD3OD:CDCl3:D2O = 50:50:1, 100 MHz), δ 175.20 (CO), 133.00 (C),

121.23 (CH), 107.36 (CH), 105.56 (CH), 70.80 (CH), 66.05 (CH2), 65.63 (CH2), 47.17

(CH2), 34.61 (CH2), 33.29 (CH2), 32.48 (CH2), 30.22 (CH2), 30.05 (CH2), 29.90 (CH2),

29.88 (CH2), 29.72 (CH2), 27.04 (CH2), 25.42 (CH2), 23.21 (CH2), 14.38 (CH3). HRMS

+ + (FAB) (m/z) calcd for C27H48NNaO9P (MNa ) 598.3121, found 598.3053.

2-(9H-Fluoren-9-ylmethoxycarbonylamino)-6-[2-[2-(9H-fluoren-9-ylmethoxy

carbonyl)-ethyl]-pyrrol-1-yl]-hexanoic acid (2.30) 6-Amino-2-(9H-fluoren-9-ylmeth-

oxycarbonylamino)-hexanoic acid (40 mg, 0.1 mmol) was suspended in 10 mL methanol

with DOHAFm (30 mg, 0.089 mmol). Then 20 μL of acetic acid was added. The

suspension dissolved gradually and a light yellow oil was generated at the bottom of the

flask as the reaction proceeded. The mixture was stirred 24 h under argon. After the

removal of solvent, the crude compound was purified by silica gel chromatography (4 %

methanol in chloroform) to give 45 mg (75 %) of a light yellow oil 2.30. TLC (4 %

1 methanol in chloroform): Rf = 0.15; H NMR (CDCl3, 400 MHz), δ 7.75 (d, J = 7.2 Hz,

4H), 7.55 (d, J = 7.6 Hz, 4H), 7.39 (dd, J = 7.2, 7.6 Hz, 4H), 7.30 (dd, J = 7.2, 7.6 Hz,

4H), 6.57 (m, 1H), 6.06 (dd, J = 3.2, 3.2 Hz, 1H), 5.87 (m, 1H), 5.4 (m, 1H), 4.41 (d, J =

6.8 Hz, 2H), 4.39 (m, 1H), 4.20 (t, J = 6.8 Hz, 2H), 3.78 (t, J = 6.8 Hz, 2H), 2.85-2.73

65 13 (4H), 1.91-1.39 (6H). C NMR (CDCl3, 100 MHz), δ 176.58 (COOH), 173.08 (COO),

156.04 (CO), 143.75 (C), 143.66 (C), 141.25 (C), 130.58 (C), 127.75 (CH), 127.69 (CH),

127.07 (CH), 127.03 (CH), 125.01 (CH), 124.95 (CH), 119.99 (CH), 106.99 (CH),

105.23 (CH), 67.04 (CH2), 66.46 (CH2), 53.49 (CH), 47.07 (CH), 46.71 (CH), 46.04

(CH2), 33.18 (CH2), 31.84 (CH2), 30.73 (CH2), 22.40 (CH2), 21.37 (CH2). HRMS (FAB)

+ + (m/z) calcd for C42H41N2O6 (MH ) 669.2959, found 669.2949.

2-(9H-Fluoren-9-ylmethoxycarbonylamino)-6-[2-[2-(9H-fluoren-9-ylmethoxy

carbonyl)-ethyl]-pyrrol-1-yl]-hexanoic acid pentafluorophenyl ester (2.31). Freshly

distilled CH2Cl2 (10 mL) was added to a mixture of pentafluorophenol (50 mg,

0.27mmol), dicyclohexylcarbodiimide (DCC, 41.5 mg, 0.201 mmol), dimethlamino pyridine (DMAP, 8 mg, 0.067 mmol) and acid 2.30 (45 mg, 0.067 mmol). The resulting mixture was stirred for 72 h at room temperature. The solvent was then removed. Flash chromatography of the residue (15 % ethyl acetate in hexane, TLC: Rf = 0.2) gave a low

1 melting white solid 2.31 (50 mg, 90 %). H NMR (CDCl3, 200 MHz) δ 7.75 (dd, J = 6.8,

6.8 Hz, 4H), 7.56 (dd, J = 7.2, 7.2 Hz, 4H), 7.38 (dd, J = 7.2, 7.6 Hz, 4H), 7.29 (4H), 6.58

(dd, J = 3.2, 1.6 Hz, 1H), 6.06 (dd, J = 3.2, 3.2 Hz, 1H), 5.87 (m, 1H), 5.38 (d, J = 8.2 Hz,

1H), 4.39-4.45 (5H), 4.17-4.22 (m, 2H), 3.84 (t, J = 7.2 Hz, 2H), 2.86 (t, J = 6.8 Hz, 2H),

13 2.77 (t, J = 6.8 Hz, 2H), 2.0-1.46 (6H). C NMR (CDCl3, 100 MHz), δ 173.14 (COO),

168.96 (COO), 156.09 (COO), 143.93 (C), 143.82 (C), 141.57 (C), 141.51 (C), 130.94

(C), 128.03 (CH), 127.32 (CH), 125.22 (CH), 120.57 (CH), 120.25 (CH), 107.41 (CH),

105.62 (CH), 67.46 (CH2), 66.73 (CH2), 53.83 (CH), 47.32 (CH), 46.99 (CH), 46.31

(CH2), 33.40 (CH2), 32.18 (CH2), 30.99 (CH2), 22.72 (CH2), 21.66 (CH2). HRMS (FAB)

+ + (m/z) calcd for C48H40F5N2O6 (MH ) 835.2801, found 835.2813.

66 Competitive ELISA for inhibition of anti-CEP antibody by CEPH-BSA. CEP-

HSA was used as a coating agent and a standard, CEPH-BSA was used as an inhibitor. A

blank, a positive control containing no inhibitor, and up to eight serial dilutions of the

inhibitor and eight serial dilutions of the CEP-HSA standard were run. Each well of a 96

well ELISA plate (Cat. 9018, Corning Inc.) was coated with CEP-HSA solution (100 μL),

prepared by diluting a solution containing 187.14 nmol/mL HSA-bound CEP in PBS to

187 pmol/mL with pH 7.4 PBS (10 mM). The plate was incubated at 37 °C for 1 h, then

washed with 10 mM PBS (3 x 300 μL), and then blocked by incubating 1 h at 37 °C with

300 μL of 1 % chicken ovalbuman (CEO) in 10 mM PBS. The plate was then rinsed with

0.1 % CEO in 10 mM PBS (300 μL). Eight serial dilutions of CEPH-BSA inhibitor or

CEP-HSA standard (120 μL each with a dilution factor of 0.2) were preincubated at 37

°C for 1 h with anti-CEP-KLH antibody solution (120 μL) that was prepared by adding 5

μL of protein G column-purified antibody (1.8 mg/mL) in PBS to 10 mL of 0.2% CEO in

10 mM PBS. The initial inhibitor and standard concentrations were 1162 pmol/mL and

935 pmol/mL, respectively. These were prepared by diluting a CEPH-BSA solution

(116.2 nmol/mL) or CEP-HSA solution (187.14 nmol/mL) with 10 mM PBS,

respectively. Blank wells were filled with 0.1 % CEO (100 μL). Positive control wells

were filled with the diluted antibody solution (50 μL) and PBS (50 μL). The antibody- antigen complex solutions (100 μL) were then added in duplicate to their respective halves of the plate, which was then incubated at room temperature with gentle agitation on a shaker for 1 h. After the supernatant was discarded, the wells were washed with 0.1

% CEO (3 x 300 μL), and then goat anti-rabbit IgG-alkaline phosphatase solution

(Boehringer-Mannheim, Indianapolis, Indiana) (100 μL) which was prepared by adding

67 10 μL of the commercial enzyme-linked secondary antibody in 10 mL of 1 % CEO was

added. The plate was then incubated at room temperature with gentle agitation for 1 h

and washed with 0.1 % CEO (3 x 300 μL). A solution of of p-nitrophenyl phosphate in

0.2 M Tris buffer (100 μL, 1.0 mg/mL, Cat. N1891, Sigma) was added. The plate was then incubated at room temperature for 20 min until the maximum absorbance reached

0.6-0.8. The development was terminated by adding 3 N NaOH (50 μL) to each well before measuring the final absorbance values. The absorbance in each well was measured with a dual-wavelength Bio-Rad 450 microplate reader with detection at 405 nm relative to 655 nm. Absorbance values for duplicate assays were averaged and scaled to make the maximum curve fit value close to 100 percent. The averaged and scaled percent absorbance values were plotted against the log of concentration. Theoretical curves for each plot were fit to the absorbance data with a four parameter logistic function, f(x) =

(a-d)/[1+(x/c)^b]+d using SigmaPlot 9.0 (Jandel Scientific Software, San Rafael, CA).

Parameter a = the asymptotic maximum absorbance, b = slope at the inflection point, c = the inhibitor concentration at the 50 % absorbance value (IC50), and d = the asymptotic minimum absorbance.

68 Table 2.2. ELISA data for CEPH-BSA of Figure 2.1.

Pmol/mL Absorbance % Absorbance Parameters Curve Fit

1162.0000 5.0000e-3 0.9634 a = 99.4463 2.6770

232.0000 0.0230 4.4316 b = 0.8436 3.9224

46.5000 0.0570 10.9827 c = 1.9337 8.4609

9.3000 0.1090 21.0019 d = 2.2389 22.6527

1.8600 0.2670 51.4451 51.6393

0.3720 0.4270 82.2736 80.0698

0.0744 0.4860 93.6416 93.5954

0.0149 0.5190 100.0000 97.8689

Table 2.3. ELISA data for CEP-HSA of Figure 2.1.

Pmol/mL Absorbance % Absorbance Parameters Curve Fit

935.0000 0.0270 3.9187 a = 102.7627 4.7109

187.0000 0.0560 8.1277 b = 0.6935 8.2860

37.4000 0.1320 19.1582 c = 3.0304 17.7498

7.4800 0.2640 38.3164 d = 2.8697 37.6602

1.4960 0.4220 61.2482 64.8032

0.2990 0.6240 90.5660 86.0684

0.0598 0.6560 95.2104 96.6024

0.0119 0.6890 100.0000 100.6648

69 2.5. References

1. Skinner, E. R.; Watt, C.; Besson, J. A.; Best, P. V., Differences in the fatty acid

composition of the grey and white matter of different regions of the brains of patients

with Alzheimer's disease and control subjects. Brain 1993, 116 ( Pt 3), 717-25.

2. Wang, N.; Anderson, R. E., Enrichment of polyunsaturated fatty acids from rat

retinal pigment epithelium to rod outer segments. Curr. Eye Res. 1992, 11, (8), 783-91.

3. Alvarez, R. A.; Aguirre, G. D.; Acland, G. M.; Anderson, R. E., Docosapentaenoic

acid is converted to docosahexaenoic acid in the retinas of normal and prcd-affected

miniature poodle dogs. Invest. Ophthalmol. Vis. Sci. 1994, 35, (2), 402-8.

4. Farnsworth, C. C.; Dratz, E. A., Oxidative damage of retinal rod outer segment

membranes and the role of vitamin E. Biochim. Biophy. Acta. 1976, 443, (3), 556-70.

5. Bok, D., New insights and new approaches toward the study of age-related macular

degeneration. Proc. Natl. Acad. Sci. U. S. A. 2002, 99, 14619-14621.

6. Ferris, F. L., 3rd Am. J. Epidem. 1983, 118, 132-151.

7. Klein, R.; Wang, Q.; Klein, B. E.; Moss, S. E.; Meuer, S. M., The relationship of

age-related maculopathy, cataract, and glaucoma to visual acuity. Invest. Ophthalmol. Vis.

Sci. 1995, 36, (1), 182-91.

8. Stone, E. M.; Sheffield, V. C.; Hageman, G. S., Molecular genetics of age-related macular degeneration. Human Molecular Genetics 2001, 10, (20), 2285-2292.

9. Williams, R. A.; Brody, B. L.; Thomas, R. G.; Kaplan, R. M.; Brown, S. I., The psychosocial impact of macular degeneration. Arch. Ophthalmol. 1998, 116, (4), 514-20.

10. Crabb, J. W.; Miyagi, M.; Gu, X. R.; Shadrach, K.; West, K. A.; Sakaguchi, H.;

Kamei, M.; Hasan, A.; Yan, L.; Rayborn, M. E.; Salomon, R. G.; Hollyfield, J. G.,

70 Drusen proteome analysis: An approach to the etiology of age-related macular

degeneration. Proc. Natl. Acad. Sci. U. S. A. 2002, 99, (23), 14682-14687.

11. Gu, X.; Meer, S. G.; Miyagi, M.; Rayborn, M. E.; Hollyfield, J. G.; Crabb, J. W.;

Salomon, R. G., Carboxyethylpyrrole protein adducts and autoantibodies, biomarkers for

age-related macular degeneration. J. Biol. Chem. 2003, 278, (43), 42027-42035.

12. Ebrahem, Q.; Renganathan, K.; Sears, J.; Vasanji, A.; Gu, X. R.; Lu, L.; Salomon,

R. G.; Crabb, J. W.; Anand-Apte, B., Carboxyethylpyrrole oxidative protein modifications stimulate neovascularization: Implications for age-related macular degeneration. Proc. Natl. Acad. Sci. U. S. A. 2006, 103, (36), 13480-13484.

13. Jackson, A. H., Pyrroles. . Comprehensive Organic Chemistry (Barton, D., and

Ollis, D. W., Eds.) 1980, 4, pp 275-320, peergamon, New York.

14. Kaur, K.; Salomon, R. G.; O'Neil, J.; Hoff, H. F., (Carboxyalkyl)pyrroles in human plasma and oxidized low-density lipoproteins. Chem. Res. Toxicol. 1997, 10, (12), 1387-

1396.

15. Sheppeck, J. E.; Kar, H.; Hong, H., A convenient and scaleable procedure for

removing the Fmoc group in solution. Tetrahedron Lett. 2000, 41, (28), 5329-5333.

16. Smith, P. K.; Krohn, R. I.; Hermanson, G. T.; Mallia, A. K.; Gartner, F. H.;

Provenzano, M. D.; Fujimoto, E. K.; Goeke, N. M.; Olson, B. J.; Klenk, D. C.,

Measurement of Protein Using Bicinchoninic Acid. Anal. Biochem. 1985, 150, (1), 76-85.

17. Bio-rad Protein Assay Instruction Manual. www.bio-

rad.com/LifeScience/pdf/Bulletin_9005.pdf

18. Decaprio, A. P.; Jackowski, S. J.; Regan, K. A., Mechanism of Formation and

Quantitation of Imines, Pyrroles, and Stable Nonpyrrole Adducts in 2,5-Hexanedione-

Treated Protein. Mol. Pharmacol. 1987, 32, (4), 542-548.

71 19. Instructions of modified Lowry protein assay kit. www.piercenet.com.

20. Sayre, L. M.; Sha, W.; Xu, G. Z.; Kaur, K.; Nadkarni, D.; Subbanagounder, G.;

Salomon, R. G., Immunochemical evidence supporting 2-pentylpyrrole formation on

proteins exposed to 4-hydroxy-2-nonenal. Chem. Res. Toxicol. 1996, 9, (7), 1194-1201.

21. Krzystolik, M. G.; Afshari, M. A.; Adamis, A. P.; Gaudreault, J.; Gragoudas, E. S.;

Michaud, N. A.; Li, W. J.; Connolly, E.; O'Neill, C. A.; Miller, J. W., Prevention of

experimental choroidal neovascularization with intravitreal anti-vascular endothelial

growth factor antibody fragment. Arch. Ophthalmol. 2002, 120, (3), 338-346.

22. Kruijff, B., Nature 1997, 386, 129-130.

23. Miyagi, M. S., H.; Darrow, R. M.; Yan, L.; West, K. A.; Aulak, K. S.; Stuehr, D. J.;

Hollyfield, J. G.; Organisciak, D. T. & Crabb, J. W., Mol. Cell. Proteomics 2002, 1, 293-

303.

24. Aulak, K. S.; Miyagi, M.; Yan, L.; West, K. A.; Massillon, D.; Crabb, J. W.; Stuehr,

D. J., Proteomic method identifies proteins nitrated in vivo during inflammatory

challenge. Proc. Natl. Acad. Sci. U. S. A. 2001, 98, (21), 12056-61.

25. West, K. A.; Yan, L.; Miyagi, M.; Crabb, J. S.; Marmorstein, A. D.; Marmorstein,

L.; Crabb, J. W., Proteome survey of proliferating and differentiating rat RPE-J cells. Exp.

Eye Res. 2001, 73, (4), 479-91.

26. Stillway, L. W.; Harmon, S. J., A procedure for detecting phosphonolipids on thin-

layer chromatograms. J. Lipid Res. 1980, 21, (8), 1141-3.

27. Crabb, J. W.; Nie, Z.; Chen, Y.; Hulmes, J. D.; West, K. A.; Kapron, J. T.; Ruuska,

S. E.; Noy, N.; Saari, J. C., Cellular retinaldehyde-binding protein ligand interactions.

Gln-210 and Lys-221 are in the retinoid binding pocket. J. Biol. Chem. 1998, 273, (33),

20712-20.

72 28. Uebel, R.; Jaarsveld, P. v.; Hawtrey, A. O., Preparation of biotinylated and the improved synthesis of some related amino derivatives. Med. Sci. Res. 1990, 18,

777-778.

73

Chapter 3

Lipid Oxidative Modifications in Autistic Disease

74 3.1. Background1, 2

In 1943, Dr. Leo Kanner, a psychiatrist at Johns Hopkins Hospital, studied a group of

11 children and applied the term ‘autism’ to children who were self-absorbed and who

had severe social, communication, and behavioral problems.3 Autism appears to affect an

estimated 34 out of every 10,000 children ages 3-10 and is three times more likely to

affect males than females.4 Autism and closely related pervasive developmental disorders

(PDDs), Asperger’s syndrome, fragile X syndrome, Rett’s syndrome and childhood

disintegrative disorder are referred to as autism spectrum disorders (ASD) that

corresponds to what the Diagnostic and Statistical Manual of Mental Disorders (DSM)-

IV refers to collectively as PDD.

Characteristics which are quite common in autism are impairments or delays in social

interaction, a lack of skills necessary for imaginative play and fantasy, and restricted,

repetitive patterns of behavior.4, 5 Autistic children do not follow the typical patterns of

child development. Some do not babble, point or make meaningful gestures by 1 year of

age. Some do not speak one word by 16 months. Some do not smile. Most children with

autism seem to have tremendous difficulty learning to participate in everyday human

interaction. They are also slower in learning to interpret what others are thinking and

feeling. Autistic patients usually exhibit odd, intensely repetitive motions although they

are physically normal and have good muscle control.

Although there is no known unique cause of autism, a number of factors have been

implicated in the pathogenesis of autism, including genetic, environmental, immunological and neurological. There is some indication of genetic influences such as

high concordance rates in siblings of autistic probands, as well as in monozygotic twins.6

75 Recently, many researchers speculate that three to five genes are likely associated with

autism.6, 7

Viruses associated with vaccinations, such as the measles component of measles-

mumps-rubella (MMR) vaccine and the pertussis component of the DPT shot were also

suspected being linked to autism.8 In the past few years, there was a public concern that the use of thimerosal (a mercury based preservative used in the MMR that is no longer used in vaccinations) linked to autism. However, a thorough review concluded that there is no causal relationship between thimerosal and autism.9, 10

Research associated with ASD

Zinc deficiency and copper excess are associated with ASD. In analyzing 503 patients

presenting diagnosis of ASD, Bill Walsh and colleagues reported 428 (85%) patients

exhibited Cu/Zn ratios in blood (1.63 ± 0.54) which were severely elevated ( p < 0.001) compared to 25 healthy controls (1.15 ± 0.24).11 Walsh proposes that copper:

imbalance leads to emotional instability, attention deficit and hyperactivity,

imbalances, and impairment of hippocampus and amygdala function.

The average autistic unbound (by ceruloplasmin) serum copper level (40 μg/dL) is 4

times higher than that of normal controls. An impaired metallothionein (MT) system is

postulated to be a pervasive factor in ASD. The absence of Cu and Zn homeostasis and

severe Zn deficiency are suggestive of an MT disorder. MTs are a family of proteins that

naturally chelate and buffer Zn as well as Cu and other redox active metals due to the

many sulfhydryl (-SH) groups they contain which give them extraordinary metal-binding

capability.12 MT synthesis and functioning involves (1) induction of thionein (apoMT), (2)

“preloading” with Zn ions, and (3) replacement of Zn by other metals, e. g., Cu. MT

76 plays a significant role in regulating the Cu levels throughout the body. It serves as a site

for temporary storage and detoxification of excess amounts of intracellular Cu.13, 14

Low plasma GSH levels are found in ASD patients. A primary mechanism for zinc loading and metal binding is the glutathione- (GSSH) redox couple.

Reduced glutathione (GSH) efficiently transfers zinc into apo-MT. Once MT is loaded with zinc, GSSH oxidizes MT causing the release of zinc in exchange for another ion, e. g., copper. Concentrations of total GSH at 4.1 ± 0.5 μmol/L were reported for autistics compared with 7.6 ± 1.4 μmol/L for healthy controls, p < 0.001.15 These abnormally low

levels of plasma GSH could contribute to the defective Cu and Zn metabolism common

in ASD patients. Furthermore, low levels of GSH also favor oxidative stress because

GSH is a for glutathione peroxidase (GPx), an antioxidant enzyme.

Sulfur metabolism defects are associated with autism. In a study of 232 children with

ASD matched with 68 control children, blood sulphate levels were found to be greatly

reduced in autism, with mean values of 0.55 nmol/mg protein compared with 4.9

nmol/mg protein for age-matched controls. In addition, urinary excretion of sulphate was

significantly elevated in autism (6819.0 ± 6712.3 nmol/mL) compared with 3030.8 ±

1461.0 nmol/mL for controls.16 Loss of sulphate in urine may partly explain low blood

sulphate levels. Sulphates in the blood help rid the body of waste peoducts by making them water soluble and therefore easily excreted. Low levels of sulphate may lead to retention of toxins, which can lead to biochemical effects on the central nervous system.

Glutathione peroxidase deficiency and superoxide dismutase excess are associated

with ASD. A major cause of damage to cells results from reactive oxygen species (ROS)

-induced alteration of macromolecules such as polyunsaturated fatty acids in membrane

77 lipids, essential proteins, and DNA. Oxidative stress and ROS have been implicated in

disease states, such as Alzheimer's disease, Parkinson’s disease, cancer, and aging. Under

normal conditions, ROS are cleared from the cell by the action of superoxide dismutase

(SOD), catalase, or glutathione peroxidase (GPx). SOD, MnSOD in the mitochondria and

CuZnSOD in the cytoplasm, removes superoxide anion by converting it into hydrogen

peroxide (H2O2). Catalase and GPx reduce H2O2 to water. In the presence of unbound Cu,

under certain conditions, SOD can promote oxidative injury owing to a Haber-Weiss

reaction of H2O2 that is catalyzed by Cu, that generates ●OH, a potent ROS. Statistically significant elevations in ZnCuSOD were documented in erythrocytes (+16.6%) and in platelets (+23.7%) of autistic individuals compared with controls, as well as severely depressed levels of GPx (-44.4%) in erythrocytes of autistic individuals compared with

17 controls. As a result, the H2O2 formed by the action of SOD would not be efficiently

removed owing to diminished levels of GPx. The imbalance is exacerbated by the low

plasma GSH levels found in ASD patients, because GSH is a cosubstrate needed for the

GPx-promoted reduction of H2O2.

Abnormalities of the immune system have been described in ASD patients. Immune

abnormalities in autism include depressed cell-mediated immunity and decreased natural

killer-cell activity;18 elevated levels of serum IgE;19 decreased proportions of interleukin-

2 (IL-2) and interferon-γ (IFN-γ) synthesizing CD4+ (T-helper-1) T cells and an increased production of IL-4 synthesizing CD4+ (T-helper-2) T cells.20 Although it is

unlikely that an immune disorder is the only or primary factor in ASD symptom

pathogenesis, the indications have led investigators to consider new multifactorial models

for the pathogenesis of autism.

78 Antibrain autoantibodies are present in the blood of ASD patients. Autoimmunity, an

abnormal immune reaction in which the immune system becomes primed to react against

body organs, can result from (1) defective “tolerance”, i. e., the absence of an immune

response to self antigens. (2) cross reactivity of self antigens with antibodies engendered

by foreign proteins. (3) an immune response against altered self proteins, e. g.., modified

by adduction of lipid-derived electrophiles generated by oxidative injury. IgG anti-brain

autoantibodies were present in 27% of sera from children with ASD compared with 2%

from healthy children. IgM autoantibodies were present in 36% of sera from children

with ASD compared with 0% of control sera.21 In another study, sera from 40 healthy

subjects and 40 autistic children were analyzed for the presence of IgG, IgM, and IgA

antibodies against nine neuron-specific antigens and three encephalitogenic and cross-

reactive proteins. Only 7.5-10% of controls had IgA, IgG or IgM antibodies against

neurofilaments compared to 37.5%, 50% and 57.5% of autistic subjects.22

The possible operation of an immune response against altered self proteins, has not been examined. Research in our group has demonstrated significantly elevated levels carboxyethylpyrrole (CEP) epitopes – specific docosahexaenoate (DHA)-derived oxidative protein modifications – and the corresponding autoantibodies in the blood of

AMD patients compared with healthy age and sex matched controls.23 Since DHA

containing lipid concentrations are uniquely high not only in retina but also in brain, it

seemed reasonable to anticipate that the detection and quantification of CEP epitopes and

autoantibodies in ASD patients will be a valuable tool for assessing oxidative injury in

ASD brain as it appears to be for assessing oxidative injury in AMD retina.

79 CEP and iso[4]LGE2-protein modifications in autistic brain. Immunohistochemical analysis of brain tissues such as cerebellum, hippocampus and neocortical regions has shown significant CEP-staining in the white matter in every autism cases but is absent in any of the control cases (age-matched or older) examined, suggesting elevated oxidative damage in these brain regions in autism (Figure 3.1).24 There is also prominent staining

24 of iso[4]LGE2-protein adduct in autistic brain but is not present in normal brain. An increased understanding of the extent and mechanisms of oxidative stress within the brain is needed, and is likely to lead to improvements in the treatment of autism. In this chapter, in order to determine whether the oxidative injury detected immunocytochemically in autistic brain was also present in blood, we examined the oxidative modifications of plasma proteins.

A B C

D E F

Figure 3.1. Anti-CEP staining of brain cortical regions from 3 of the control cases aged

5, 6 and 11 years (A, B, C) and 3 autism cases aged 9, 7, and 5 years (D, E, F).

80 In the present study, levels of CEP and iso[4]LGE2 adducts in plasma from patients

with documented ASD were compared with age-matched healthy controls. Anti-CEP,

anti-iso[4]LGE2, autoantibodies were also measured. In addition, since recent studies

proposed a possible participation of enhanced nitrative stress in AMD pathologies,25 levels of protein bound nitrotyrosine in plasma from ASD and normal controls were compared in this study by stable isotope dilution tandem mass spectrometry. No significant elevations in the levels of any of these markers of oxidative injury were detected in ASD plasma. However, an unanticipated strong elevation of iso[4]LGE2 immunoreactivity levels were found in ASD patients born prematurely compared with

ASD patients with no birth events or other birth events.

81 3.2. Materials and Methods

Human plasma

Blood samples were obtained by phlebotomists at the Institute’s Pfeiffer Treatment

Center. Eligibility for the study was based on a diagnosis of ASD, as defined by DSM-IV.

Subjects with questionable diagnoses and patients with comorbidity for seizures, and

birth anoxia were excluded. In total, 27 autistic subjects and 11 healthy control subjects

were recruited for the study.

ELISA

A competitive enzyme-linked immunosorbent assay (ELISA) of plasma from ASD and

healthy controls was performed as described previously for CEP and iso[4]LGE2

26 23 immunoreactivities. A direct ELISA was performed for anti-CEP and anti-iso[4]LGE2

autoantibodies. Absorbance values were measured on a Bio Rad Microplate Reader using

dual wavelength (405 nm to read the plate and 650 nm as a reference).

Mass spectrometric analysis of nitrotyrosine

Protein-bound nitrotyrosine in plasma from ASD and healthy controls was quantified

by stable isotope dilution LC/MS/MS as described27 on a triple quadrupole mass

spectrometer (API 365, Applied Biosystems, Foster City, CA) with Ionic EP 10+ upgrade

(Concord, Ontario, CA) interfaced to a Cohesive Technologies Aria LX Series HPLC

multiplexing system (Franklin, MA). Samples were delipidated, hydrolyzed, passed over mini solid-phase C18 extraction columns (Supelclean LC-C18-SPE minicolumn, 3 mL,

Supelco, Inc., Bellefone, PA) and subjected to mass spectrometric analysis. Synthetic

13 [ C6]-labeled 3-nitrotyrosine was used as internal standard for quantification of natural abundance 3-nitrotyrosine. Simultaneously, a universal labeled precursor amino acid,

82 13 15 [ C9, N1]tyrosine, was added, permitting potential intrapreparative formation of

13 15 nitro[ C9, N1]tyrosine to be routinely monitored and shown to be negligible (i.e., <<

5% of the level of the natural abundance product observed). Results are normalized to the content of the precursor amino acid tyrosine (i.e., nitrotyrosine/tyrosine, micromoles/ moles), which was monitored within the same injection.

Statistical analyses

ASD and normal controls were compared using CEP, iso[4]LGE2 protein adducts, anti-

CEP, anti-iso[4]LGE2 antibodies and nitroTyr/Tyr ratio. P-values were calculated by

independent t-test and analysis of variance (ANOVA) using Microsoft Excel 2003 for

Windows. The significance of group comparisons was corrected for the effect of repeated measures. Correlations between measured parameters were ascertained using pair-wise

comparisons by linear regression and were performed using JMP 5.12 (SAS Institute,

Cary NC). Data were expressed as mean ± SD. Statistical significance was defined as p less than 0.05.

83 3.3. Results and Discussion

3.3.1. CEP and iso[4]LGE2 protein adducts immunoreactivity in human plasma.

Levels of CEP and iso[4]LGE2-protein adducts in human blood were measured by

competitive ELISA using polyclonal anti-CEP and anti-iso[4]LGE2-KLH antibodies

respectively. We examined plasma from 27 patients with diagnosed ASD and 11 healthy

controls with matched ages. Comparison of levels of CEP-protein adducts in ASD and

controls is presented in Figure 3.2. Levels of individual CEP-protein adducts are shown

in Figure 3.3.

CEP Immunoreactivity in ASD and Control 350

p = 0.45 300

250

200

150 125 ± 58 110 ± 30 100

CEP Immunoreactivity (pmol/mL) 50

0 Autism Control (n = 27) (n = 11)

Figure 3.2. CEP immunoreactivity detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set.

84 CEP Immunoreactivity of ASD and Controls

350

300

250 ASD Control 200

150

100

50 CEP Im m unoreactivity (pm ol/m L)

0

2 4 6 7 9 8 3 1 4 6 9 A1 A A3 A A5 A A A8 A 10 11 12 14 16 17 19 21 22 24 26 27 N N2 N3 N N5 N N7 N8 N 11 A A A A13 A A15 A A A1 A A20 A A A2 A A25 A A N10 N Sample ID Figure 3.3. Levels of individual CEPs in plasma from 27 patients with diagnosed ASD and 11 healthy controls.

Comparison of levels of iso[4]LGE2-protein adducts is presented in Figure 3.4. Levels

of individual iso[4]LGE2-protein adducts are shown in Figure 3.5.

Iso[4]LGE2 Immunoreactivity in ASD and Control 40 p = 0.09 35

30

25

20 16.7 ± 5.8 15 Immunoreactivity (nmol/mL)

2 13.4 ± 3.4

10

iso[4]LGE 5 Autism Control (n = 27) (n = 11)

Figure 3.4. Iso[4]LGE2 immunoreactivity detected in human plasma from (◆) ASD and

(■) normal controls. The figure also shows mean levels detected (O). The error bars

indicate the standard deviation (S. D.) for each data set.

85 Iso[4]LGE2 Immunoreactivity of ASD and Control

35

30 ASD Control 25

20

15 Immunoreactivity 2 (nmol/mL) 10

5 Iso[4]LGE

0

3 4 5 6 7 2 3 4 5 2 3 4 5 6 7 8 9 A1 A2 A A A A A A8 A9 1 1 1 1 16 2 2 2 2 N1 N2 N3 N4 N5 N N N N A10A11A A A A A A17A18A19A20A21A A A A A26A27 N10N11 Sample ID

Figure 3.5. Levels of individual iso[4]LGE2-protein adducts in plasma from 27 patients

with diagnosed ASD and 11 healthy controls.

A typical inhibition curve for binding of iso[4]LGE2-KLH with a naturally occurring

epitope in plasma from a patient with documented ASD and a normal control is shown in

Figure 3.6.

120

100

80

60

40

Absorbance (% max) Absorbance (% 20

0

10-3 10-2 10-1 100 101 102 103 104 Inhibitor (nmol/mL)

Figure 3.6. Inhibition curves for binding of anti-iso[4]LGE2-KLH to iso[4]LGE2-BSA by

iso[4]LGE2-HSA (●), plasma from an ASD patient (■), and a normal control (▲).

86 There were no significant differences in the mean levels of CEP adduct between the

plasma of ASD, 124.5 ± 57.9 pmol/mL, and the plasma of age-matched healthy

controls, 110.4 ± 30.3 pmol/mL, p = 0.45. Nor were there statistically significant

differences in the mean levels of iso[4]LGE2 protein adduct between ASD, 16.7 ± 5.8

nmol/mL, and controls, 13.4 ± 3.4 nmol/mL, p = 0.088.

3.3.2. Presence of anti-CEP and anti-iso[4]LGE2 autoantibodies in human plasma.

ASD seems to arise from environmental factors interacting with a genetic predisposition. It often occurs in conjunction with a family history of autoimmune

diseases.28-30 As mentioned above, autistic patients often have immune abnormalities.

Antibrain antibodies IgG, IgM, IgE and IgA are present in ASD patients.21 Inflammatory

over activation is suggested by the observation that many autistic children appear to

produce excessive amounts of TNFα and other pro-inflammatory cytokines.31 In this

study, we were interested in probing the association between ASD and lipid oxidation.

Anti-CEP autoantibody titer in plasma from 26 ASD and 5 healthy controls were

measured by ELISA. Data obtained from triplicate assays are summarized in Figures 3.7

and 3.8. Anti-iso[4]LGE2 autoantibody titer in plasma of 27 ASD and 11 healthy

controls are summarized in Figures 3.9 and 3.10.

87 CEP Autoantibody Titer in ASD and Control 4 p = 0.9 ) 0

3

2

1.2 ± 0.7 1.2 ± 0.3 1 CEP Autoantibody Titer (A/A Titer CEP Autoantibody

0 Autism Control (n = 26) (n = 5)

Figure 3.7. CEP autoantibody titer detected in human plasma from (◆) ASD and (■)

normal controls. The figure also shows mean levels detected (O). The error bars indicate

the standard deviation (S. D.) for each data set.

CEP Autoantibody Titre of ASD and Control

4.0 3.5 3.0 ASD Control 2.5 2.0 1.5 1.0 0.5

CEP Autoantibody Titre(A/A0 0.0

1 4 6 9 0 5 0 2 5 2 3 5 A A2 A3 A A5 A A7 A8 A 13 14 19 24 N1 N N N4 N A1 A11A12A A A1 A16A17A18A A2 A21A2 A23A A2 A26 Sample ID

Figure 3.8. Levels of individual individual CEP autoantibody titer in plasma from 26 patients with diagnosed ASD and 5 healthy controls.

88 Iso[4]LGE2 Autoantibody Titer in ASD and Control

8

) p = 0.6 7

6

5

4

3

Autoantibody Titer (A/A0 Autoantibody 2 2 1.3 ± 1.6 1 1.0 ± 0.9

0 Iso[4]LGE

-1 Autism Control (n = 27) (n = 11)

Figure 3.9. Iso[4]LGE2 autoantibody titer detected in human plasma from (◆) ASD and

(■) normal controls. The figure also shows mean levels detected (O). The error bars

indicate the standard deviation (S. D.) for each data set.

Iso[4]LGE2 Autoantibody Titer 9

8

7 ASD Control 6

5 4

3 Autoantibody Titer (A/A0) Titer Autoantibody 2 2 1

Iso[4]LGE 0

3 6 9 2 6 9 3 6 3 4 6 7 A1 A2 A A4 A5 A A7 A8 A 10 1 13 1 17 1 20 2 24 2 27 N1 N2 N N N5 N N N8 N9 10 A A11A A A14A15A A A18A A A21A22A A A25A A N N11 Sample ID

Figure 3.10. Levels of individual iso[4]LGE2 autoantibody titer in plasma from 27

patients with diagnosed ASD and 11 healthy controls.

89 There were no significant differences in the mean levels of anti-CEP autoantibody

titer between the plasma of ASD, 1.2 ± 0.7, and the plasma of age-matched healthy

controls, 1.2 ± 0.3, p = 0.9. Nor were there statistically significant differences in the

mean levels of anti-iso[4]LGE2 autoantibody titer between ASD, 1.3 ± 1.6, and

controls, 1.0 ± 0.9, p = 0.6.

3.3.3. Protein bound nitrotyrosine, chlorotyrosine and bromotyrosine levels in ASD and healthy controls.

A number of inflammatory and neurodegenerative disorders have been linked with protein modification by reactive nitrogen species including ocular inflammation, atherosclerosis, retinal ischemia, lung infection, cancer, Parkinson’s disease, and

32 Alzheimer’s disease. Measurement of NO2Tyr, a posttranslational modification of proteins generated by reactive nitrogen species, serves as a quantitative index of nitrative stress in vivo.

In this study, there were no differences between the ratio of NO2Tyr/Tyr in plasma

from 27 ASD patients, 7.81 E -06 ± 3.29 E -06, and that from 11 healthy controls, 7.87

E-06 ± 1.62 E -06. The data are shown in Figure 3.11.

We also measured chlorotyrosine and bromotyrosine levels in autistic and normal plasma.

There were no differences between the ratio of ClTyr/Tyr in plasma from 27 ASD

patients, 1.37 E -05 ± 1.03 E -05, and that from 11 healthy controls, 1.55 E -05 ± 1.14

E-05. The data are shown in Figure 3.12. Nor were there any significant differences

between the ratio of BrTyr/Tyr in plasma from ASD patients, 4.17 E -05 ± 5.69 E -06,

and that from healthy controls, 3.77 E -05 ± 7.41 E -06. The data are shown in Figure

3.13.

90 NitroTyr/Tyr in Autism and Control Plasma 2.00E-05 p = 0.95 1.80E-05

1.60E-05

1.40E-05

1.20E-05

1.00E-05

8.00E-06 7.81 ± 3.29 7.87 ± 1.62 6.00E-06

4.00E-06

2.00E-06 NitroTyr/Tyr Conc. Ratio (mol/mol) Ratio Conc. NitroTyr/Tyr 0.00E+00 Autism Control (n = 27) (n = 11)

Figure 3.11. Nitrotyr/Tyr ratio detected in human plasma from (◆) ASD and (▲) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set.

ClTyr/Tyr in Autism and Control Plasma

4.50E-05 p = 0.59 4.00E-05

3.50E-05

3.00E-05

2.50E-05

2.00E-05

1.50E-05 1.55 ± 1.14 1.37 ± 1.03 1.00E-05 ClTyr/Tyr Conc. Ratio (mol/mol) Ratio Conc. ClTyr/Tyr 5.00E-06

0.00E+00 Autism Control (n = 27) (n = 11)

Figure 3.12. Cltyr/Tyr ratio detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set.

91 BrTyr/Tyr in Autism and Control Plasma

7.00E-05 p = 0.08 6.50E-05

6.00E-05

5.50E-05

5.00E-05

4.50E-05 4.17 ± 0.57 4.00E-05 3.77 ± 0.74 3.50E-05 BrTyr/Tyr Conc. Ratio (mol/mol) Ratio Conc. BrTyr/Tyr 3.00E-05

2.50E-05 Autism Control (n = 27) (n = 11)

Figure 3.13. Brtyr/Tyr ratio detected in human plasma from (◆) ASD and (■) normal controls. The figure also shows mean levels detected (O). The error bars indicate the standard deviation (S. D.) for each data set.

3.3.4. Correlations among lipid oxidative protein adducts and nitrative proteins.

Linear regression analyses of CEP, iso[4]LGE2 immunoreactivities and nitroTyr/Tyr

were conducted (Figures 3.14-3.17). Out of 27 ASD patients, 6 (22%) showed elevated

CEP and iso[4]LGE2 immunoreactivities while 1 out of 11 normal controls (9%) showed

both elevated CEP and iso[4]LGE2 immunoreactivities (Figure 3.15 ).

A significant linear correlation, R = 0.34, p = 0.036, was found between the levels

of CEP and iso[4]LGE2 immunoreactivities (Figure 3.14, non-parametric Spearman’s

correlation was used). There were no significant linear correlations between CEP and

nitroTyr/Tyr (Figure 3.16), R = 0.09, p = 0.56, or iso[4]LGE2 immunoreactivity and

nitroTyr/Tyr (Figure 3.17), R = 0.1, p = 0.5.

92 300

250

200 ● ASD (n = 27) ▲ Control (n = 11) 150 R = 0.34, p = 0.036

100

50 CEP immunoreactivity (pmol/mL) 10 15 20 25 30

Iso[4]LGE2 immunoreactivity (nmol/mL)

Figure 3.14. Correlation between CEP immunoreactivity and iso[4]LGE2

immunoreactivity in human plasma from (●) ASD and (∆) normal controls.

Correlation of Iso[4]LGE2 and CEP Immunoreactivity 350.0

III 300.0 IV Š ASD (n = 27) 250.0 ƒ Control (n = 11) c Ave. 200.0

eactivity (pmol/mL) 150.0

100.0 II 50.0 I CEP Immunor CEP

0.0 5.0 10.0 15.0 20.0 25.0 30.0 35.0

Iso[4]LGE2 Immunoreactivity (nmol/mL)

Figure 3.15. Correlation between iso[4]LGE2 and CEP immunoreactivities in human plasma from (◆) ASD and (■) normal controls. The horizontal and vertical dashed lines

indicate the mean values of CEP and iso[4]LGE2 immunoreactivities, respectively. Out of

27 ASD patients, 6 (22%) are in region IV while 1 out of 11 normal controls (9%) is in region IV. ASD patients account for 86% (6/7) of the data in region IV.

93 300

250

200 ● ASD (n = 27) ▲ Control (n = 11) CEP 150 R = 0.09, p = 0.56

immunoreactivity 100

50

0 5 10 15 20 Nitrotyro/Tyr

Figure 3.16. Correlation between CEP immunoreactivity and nitrotyro/Tyr ratio in

human plasma from (●) ASD and (∆) normal controls.

30

25 ● ASD (n = 27) y

▲ Control (n = 11) 20 R = 0.1, p = 0.5 immunoreactivity 2

(nmol/mL) 15

10 Iso[4]LGE

0 5 10 15 20 Nitrotyro/Tyr

Figure 3.17. Correlation between iso[4]LGE2 immunoreactivity and nitrotyro/Tyr ratio in human plasma from (●) ASD and (∆) normal controls.

3.3.5. Correlations between autoantibodies and lipid oxidation immunoreactivities.

Although CEP autoantibody titer was only monitored in plasma from 26 ASD and 5

controls, a strong correlation was seen in CEP autoantibody titer and iso[4]LGE2

autoantibody titer (Figure 3.18), R = 0.76, p < 0.001.

94 No correlation was found between CEP imunoreactivity and CEP autoantibody levels

(Figure 3.19), R = 0.28, p = 0.6. Nor was there any correlation between iso[4]LGE2 immunoreactivity and iso[4]LGE2 autoantibody levels (Figure 3.20), R = 0.04, p = 0.8.

3.5

3

2.5 ● ASD (n = 26) ▲ Control (n = 5) 2

titer R = 0.76, p < 0.001

1.5 CEP autoantibody

1

0.5 0 1 2 3 4 5 6 7 8

Iso[4]LGE autoantibody titer 2

Figure 3.18. Correlation between CEP autoantibody titer and iso[4]LGE2 autoantibody

titer in human plasma from (●) ASD and (∆) normal controls.

300

250

200 ● ASD (n = 26) ▲ Control (n = 5) CEP 150 R = 0.28, p = 0.6

immunoreactivity 100

50

.5 1 1.5 2 2.5 3 3.5 CEP autoantibody titer

Figure 3.19. Correlation between CEP immunoreactivity and CEP autoantibody titer in

human plasma from (●) ASD and (∆) normal controls.

95 30

25

)

20 ● ASD (n = 27) ▲ Control (n = 11)

immunoreactivity R = 0.04, p = 0.8 2 nmol/mL ( 15

Iso[4]LGE 10

0 1 2 3 4 5 6 7 8

Iso[4]LGE autoantibody titer 2

Figure 3.20. Correlation between iso[4]LGE2 immunoreactivity and iso[4]LGE2 autoantibody titer in human plasma from (●) ASD and (∆) normal controls.

3.3.6. Lipid oxidation products and birth events in ASD patients.

Figures 3.21-24 show differences in levels of lipid oxidation protein adducts and autoantibody titers, grouped according to birth events of ASD patients with no events, born premature or other events such as C-section, cord wrapped and low birth weight.

The “born premature" group exhibits a significantly elevated mean level of

iso[4]LGE2 immunoreactivity, compared to “no birth events” group (p = 0.008) and

the “other events” group (p = 0.016). However, neither CEP immunoreactivity level

nor CEP or iso[4]LGE2 autoantibody titers showed any significant differences.

It must be noted that all of the “born premature” group were also a cohort of the

autistic group. Thus, we had no “born premature” individuals in the control group. So it is

not possible to distinguish between a general characteristic of premature birth and a

characteristic of a subgroup of autistic patients.

96 Birth Events and CEP Immunoreactivity 255

p = 0.88 p = 0.23 205

155

128 ± 34 124 ± 59 105 102 ± 32

55

5 CEP Immunoreactivity (pmol/mL) Immunoreactivity CEP No Significant Born Other Events Birth Events Premature (n=5) (n=16) (n=6)

Figure 3.21. CEP immunoreactivity grouped according to birth events of ASD patients with no events, born premature or other birth events.

Birth Events and CEP Autoantibody Titer 4.0 p = 0.8 p = 0.9 3.5

3.0

2.5

2.0

1.5 1.2 ± 0.8 1.1 ± 0.7 1.2 ± 0.7 1.0

0.5 CEP autoantibody titer (A/A0) titer CEP autoantibody

0.0 No Significant Born Other Events Birth Events Premature (n=5) (n=16) (n=5)

Figure 3.22. CEP autoantibody titer grouped according to birth events of ASD patients with no events, born premature or other birth events.

97 Birth events and iso[4]LGE2 immunoreactivity 35 p = 0.008 p = 0.016

30

25 22.7 ± 5.9 20

15.3 ± 5.0 15 14.0 ± 3.0 immunoreactivity (nmol/mL) 2

10

Iso[4]LGE 5 No Significant Born Other Events Birth Events Premature (n=5) (n=16) (n=6)

Figure 3.23. Iso[4]LGE2 immunoreactivity grouped according to birth events of ASD patients with no events, born premature or other birth events.

Birth Events and iso[4]LGE Autoantibody Titer 8.5 2

7.5 p = 0.8 p = 0.98 6.5

5.5

4.5

3.5

2.5 autoantibody titer (A/A0) 2 1.5 1.4 ± 1.6 1.5 ± 1.3 1.2 ± 1.8 0.5 iso[4]LGE -0.5 No Significant Born Other Events Birth Events Premature (n=5) (n=16) (n=6)

Figure 3.24. Iso[4]LGE2 autoantibody titer grouped according to birth events of ASD patients with no events, born premature or other birth events.

98 3.4. Conclusions

In view of the high omega-3 poly unsaturated fatty acid content of the brain, it is likely

that these fats participate in brain biochemistry, physiology and functioning; and may

play a role in some neuropsychiatric diseases and ageing. By using polyclonal anti-

iso[4]LGE2 and anti-CEP antibodies, we measured levels of the lipid oxidation protein adducts iso[4]LGE2 and CEP in blood plasma from 27 autistic patients and 11 normal controls. Autoantibodies against iso[4]LGE2 and CEPs were also monitored by direct

ELISA. Similar levels of these protein modifications and autoantibodies against them

were detected in blood from both autistic individuals and normal controls.

Immunohistochemically, it was discovered that modifications of brain protein by

products of free radical-induced lipid oxidation, i.e., CEP and iso[4]LGE2-protein adducts,

are uniquely present in autistic brain but not in normal brain. Taken together, our

observations suggest that the oxidative protein modifications associated with autistic

brain are highly localized and do not cross the blood/brain barrier.

Unexpectedly, a subpopulation consisting of autistic patients born prematurely showed significantly elevated iso[4]LGE2-protein adduct immunoreactivity levels compared with patients with no birth events or other birth events. No control subjects were born

prematurely in this study, so it can not be concluded whether or not this phenomenon is associated with autism. The possibly general association of the phenomenon of elevated plasma iso[4]LGE2-protein adduct levels does warrant further investigation as a

heretofore unrecognized consequence of premature birth or postnatal treatment protocols.

Other observations, that are not associated with autism, are strong correlations between

CEP and iso[4]LGE2-protein adduct immunoreactivity levels in plasma from 27 ASD

99 patients and 11 normal controls, as well as between anti-CEP and anti-iso[4]LGE2- protein adduct autoantibody titers in plasma from 26 ASD patients and 5 normal controls.

The positive correlation between the two lipid oxidation protein modifications suggests that oxidative damage to docosahexaenoate and arachidonate are equally likely. Also, the proclivity of an individual to develop an immune response to altered self protein is similar for a diverse structural array of immunogenic modifications, e. g., CEPs and isoLGs.

Tandem mass spectrometry was used to monitor the levels of protein nitrative products

(nitrotyrosine) in plasma from 27 ASD and 11 normal controls. We found no significant differences between ASD and controls.

100 3.5. Experimental Procedures

Human plasma

Plasma samples were treated according to following protocol:

1. Collect 5 mL of blood in 7 mL lavender top vacutainer tube with EDTA (chilled tube

is not necessary). Gently invert the tube 5-6 times to distribute anticoagulant.

2. Spin in refrigerated centrifuge (4 °C) at 3000 rpm for 15 minutes.

3. Transfer plasma (2.0 mL, be sure to avoid the buffy coat) to a 15 mL Falcon tube.

4. Add 10 μL of BHT/ethanol solution (5 μL/mL plasma) and 20 μL of protease inhibitor

cocktail kit (10 μL/mL plasma, Sigma, Cat. P8340) to plasma. Mix gently 5-6 times by

inversion. Do not vortex. The BHT/ethanol solution is made by combining BHT (44 mg)

with ethanol (10 mL). If refrigerated, the BHT solution is good for 3 weeks.

5. Aliquot 250 μL into 8 prelabeled screw-top microfuge tubes. Gently flush the vials

with argon and seal with screw caps.

6. Quench freeze in liquid nitrogen by placing the 8 vials into a Scienceware Round

Bubble Rack (Fisher, Cat. 14-792-14 Bel-Art, fishersci.com) and then immersing the

bottom of the rack and the tubes into liquid nitrogen in Nalgene polyethylene Dewar flask

for 1 minute.

7. Store vials in fiberboard storage boxes (Fisher, Cat. 11-678-24A, fishersci.com) with fiberboard box dividers (Fisher, Cat. 11-678-24C, fishersci.com) in dry ice for transport

or at -80 °C.

101 Biomarkers

CEP-BSA and CEP-HSA were prepared as described in Chapter 2. Iso[4]LGE2-BSA and iso[4]LGE2-HSA were prepared as follows: BSA (20.6 mg) in pH = 7.4 PBS solution

(10 mM, 2 mL) was incubated with iso[4]LGE2 (3 mg) at 37 °C for 20 h under Argon.

The light yellow clear solution changed to green-yellow clear solution as the reaction

going. The consequent iso[4]LGE2-BSA was diluted with PBS (10 mM, 3 mL) and

transferred to a dialysis membrane tube ( Mr cutoff 14000). Two successive dialyses (12

h each) against 1 L 10 mM PBS (pH 7.4) at 4 °C were performed. The final protein

concentration (3.745 mg/mL) was determined by the Pierce bicinchoninic acid (BCA)

protein assay.33 The pyrrole concentration (1.094 mM) was determined by Ehrlich

34 assay. The pyrrole : protein ratio was found to be 11 : 1. Iso[4]LGE2-HSA was

synthesized similarly except that it was diluted with 4 mL PBS (10 mM). Protein concentration was 2.82 mg/mL by the Pierce bicinchoninic acid (BCA) protein assay33,

and pyrrole concentration was 0.842 mM by Ehrlich assay.34 The pyrrole : protein ratio

was also found to be 11 : 1. Anti-CEP-KLH, anti-iso[4]LGE2-KLH polycolonal

antibodies were purified as described previously.23, 26

Competitive ELISA for CEP and iso[4]LGE2 immunoreactivity. ELISA of plasma

from ASD and healthy controls was performed as described in Chapter 2. For CEP immunoreactivity, CEP-BSA was used as a coating agent and CEP-HSA was used as a standard. The initial concentration of CEP-HSA was 16.7 nmol/mL. For iso[4]LGE2 immunoreactivity, iso[4]LGE2-BSA was used as a coating agent and iso[4]LGE2-HSA was used as a standard. The initial concentration of iso[4]LGE2-HSA was 810 nmol/mL.

A dilution factor of 0.2 was employed for standard and samples. Eight serial dilutions for

102 standard, five serial dilutions of samples were performed. Data for Figure 3.6 are presented in Tables 3.1-3.3. Levels of CEP and iso[4]LGE2 immunoreactivities in plasma

of ASD patients versus age matched healthy control are summarized in Table 3.4-3.5.

Table 3.1. ELISA data for iso[4]LGE2-HSA standard for Figure 3.6.

nmol/mL Absorbance % Absorbance Parameters Curve fit

810.0000 0.0100 1.4493 98.9827 1.6297

162.0000 0.0200 2.8986 1.2176 2.4593

32.4000 0.0540 7.8261 3.6946 7.9634

6.4800 0.2330 33.7681 1.4923 34.1853

1.3000 0.5440 78.8406 77.6367

0.2590 0.6350 92.0290 95.2946

0.0520 0.6920 100.2899 98.4430

0.0100 0.6860 99.4203 98.9098

Table 3.2. ELISA data for ASD patient (patient code A27) for Figure 3.6.

nmol/mL Absorbance % Absorbance Parameters Curve fit

810.0000 0.1350 17.9045 98.4569 19.3619

162.0000 0.4080 54.1114 0.9558 52.4454

32.4000 0.6180 81.9629 185.7748 82.8472

6.4800 0.6960 92.3077 1.7911e-7 94.6278

1.3000 0.7540 100.0000 97.6062

103 Table 3.3. ELISA data for control (normal code N11) for Figure 3.6.

nmol/mL Absorbance % Absorbance Parameters Curve fit

810.0000 0.1250 17.3130 99.2831 17.9040

162.0000 0.4790 66.3435 1.3534 65.5440

32.4000 0.6640 91.9668 264.6144 93.8141

6.4800 0.7160 99.1690 1.5995e-7 98.6319

1.3000 0.7220 100.0000 99.2086

CEP and iso[4]LGE2 autoantibodies in human plasma.

Direct ELISAs were performed to determine the antibody titer. The plates were coated with CEP-BSA or iso[4]LGE2-BSA (100 μL/well). BSA (2 %) in 10 mM PBS (100 μL) was added to the corresponding blank wells. The plates were incubated for 1 h at 37 °C, washed with PBS (10 mM, 300 μL) 3 times, and blocked with 1% chicken egg ovalbumin (CEO, 300 μL) for 1 h at 37 °C. After washing once with 0.1 % CEO plus

0.05 % Tween 20 (300 μL), the plates were loaded with plasma, diluted 20 times in 0.2 %

CEO plus 0.05 % Tween 20, and then incubated for 1 h at room temperature. The plates were then washed 3 times with 0.1 % CEO plus 0.05 % Tween 20 (300 μL), and then incubated 1 h at room temperature with alkaline phosphatase conjugated goat anti-human

IgG (Sigma-Aldrich, Milwaukee, WI, Cat. Sigma A-8542), which was diluted 1:2000 with 1% CEO plus 0.05 % Tween 20 (100 μL). After washing with 0.1% CEO (3 × 300

µL), a solution of p-nitrophenyl phosphate in 0.2 M Tris buffer (1.0 mg/mL, 100 µL,

Sigma-Aldrich, Milwaukee, WI, Cat. Sigma N1891) was added. The absorbance was read at 405 nm with reference at 650 nm after incubation at room temperature for 30 min. The

104 titer was defined as the ratio of plasma binding to antigen (A) vs. binding to BSA blank

(A0). The data for CEP and iso[4]LGE2 autoantibodies are list in Tables 3.4 and 3.5.

Table 3.4. Data for CEP, iso[4]LGE2 immunoractivites and CEP, iso[4]LGE2 autoantibodies in ASD patients.

CEP iso[4]LGE2 CEP iso[4]LGE2

Patient code immunoreactivity immunoreactivity autoantibody autoantibody

(pmol/mL) (nmol/mL) (A/A0) (A/A0)

A1 60.8 8.8 0.9 1.1

A2 105 14.4 1.1 0.6

A3 143.2 15.2 0.9 0.4

A4 215.6 14 3.4 2.1

A5 55.2 14 2.8 7.7

A6 87.1 12.8 2.2 3.6

A7 185 20 0.7 0.3

A8 102.5 24.8 1.1 1.4

A9 167.5 9.6 0.7 0.5

A10 172 21.6 0.9 0.8

A11 287.5 23.2 1.5 1

A12 237.5 13.6 1.1 1.2

A13 93.2 12 1.6 1.8

A14 135.7 29.6 2.4 4.5

A15 158.8 22.4 0.6 0.5

A16 79.4 22.8 0.8 0.6

105 A17 104 12 0.7 0.9

A18 91.5 14.8 0.9 0.7

A19 154.6 19.2 0.7 0.3

A20 72.8 14 0.6 0.7

A21 41.7 9.6 1.2 1.3

A22 84.3 13.6 0.7 0.2

A23 112.5 16 0.8 0.4

A24 100 29.6 1 0.8

A25 115.3 10.4 0.9 0.4

A26 87.6 16.8 0.7 0.5

A27 111.2 16.2 . 1.5

Table 3.5. Data for CEP, iso[4]LGE2 immunoractivites and CEP, iso[4]LGE2 autoantibodies in normal controls.

CEP iso[4]LGE2 CEP iso[4]LGE2

Normal code immunoreactivit immunoreactivity autoantibody autoantibody

y (pmol/mL) (nmol/mL) (A/A0) (A/A0)

N1 102.1 10 1.6 0.4

N2 130.1 9.6 1.4 1.1

N3 182.5 19.2 0.8 0.3

N4 70.5 9.6 1.1 0.6

N5 98.6 16.8 1.2 0.4

N6 111.9 13.1 3.2

106 N7 83.6 13.1 0.5

N8 91.8 10.5 1.5

N9 97.4 16.9 1.7

N10 111.6 16.2 0.4

N11 134.5 12.8 1.2

Detection of nitrotyrosine in plasma of ASD and control. This work was done

collaboratively with Xiaoming Fu and Zeneng Wang.35

Protein Hydrolysis—Protein was delipidated and desalted using two sequential

extractions with a single phase mixture of H2O/methanol/H2O-saturated

13 13 (1:3:8 v/v/v). Oxidized tyrosine standards, C6-ortho-tyrosine (o-Tyr), C6-meta-

13 13 13 tyrosine (m-Tyr), C6-nitrotyrosine (Nitro-Tyr), C6-chlorotyrosine (Cl-Tyr), C6-

13 15 bromotyrosine (Br-Tyr) (2 pmol each) and universal labeled tyrosine, C9- N-tyrosine

(2 nmol) were added to protein pellets. Samples were hydrolyzed in degassed 4M

methane sulfonic acid (500 μL) supplemented with 1% phenol for 24 h at 110 °C under

argon atmosphere. Amino acid hydrolysates were resuspended in 0.1% trifluoroacetic acid (2 mL) and applied to mini solid-phase C18 extraction columns (Supelclean LC-C18

SPE mini-column, 3 ml; Supelco, Inc., Bellefone, PA) pre-equilibrated with 0.1% trifluoroacetic acid. Following sequential washes with trifluoroacetic acid (2 ml, 0.1%), oxidized tyrosines and tyrosine were eluted with 2 ml of 30% methanol in 0.1% trifluoroacetic acid, dried under vacuum, and then analyzed by mass spectrometry as described below.

107 Mass Spectrometry (MS)—Nitrotyrosine in lysates was analyzed by HPLC with on-line

electrospray ionization tandem mass spectrometry (LC/ESI/MS/MS) using stable isotope

dilution methodology on a triple quadrupole mass spectrometer (API 365; Applied

Biosystems, Foster City, CA) with Ionics EP 10+ upgrade (Ionics, Concord, Ontario, CA)

interfaced to a Cohesive Technologies (Franklin, MA) Aria LX Series HPLC

multiplexing system. Samples were suspended in equilibration solvent (H2O with 0.1%

formic acid) and injected onto an Ultrasphere C18 column (Phenominex, 5 μm, 2.0 × 150

mm). L-Tyrosine and its oxidation products were eluted at a flow rate of 0.2 mL/min

using a linear gradient generated against 0.1% formic acid in acetonitrile pH 2.5, as the

second mobile phase. Analytes were monitored in positive ion mode with full scan

product ion MS/MS at unit resolution. Response was optimized with a spray voltage

setting of 5 kV. The heated capillary voltage was set at 10 V and the temperature to 350

°C. Nitrogen was used both as sheath and auxiliary gas, at a flow rate of 12 and 8 psi,

respectively. The precursor ion isolation width was 1.0 and 3.0 for nitrotyrosine and

tyrosine, respectively. The analyte abundance was evaluated by measuring the

chromatographic peak areas of selected product ions extracted from the full scan total ion

chromatogram, according to the corresponding ion trap product ion spectra. The ions

12 monitored for each analyte were: 3-nitro[ C6]tyrosine (mass-to-charge-ratio (m/z)

13 13 15 227→181); 3-nitro[ C6]tyrosine (m/z 233→187); 3-nitro[ C9 N1]tyrosine (m/z

35 12 35 13 237→190); chloro-[ C6]tyrosine (m/z 216→170); chloro-[ C6]tyrosine (m/z

35 13 15 12 222→176); chloro-[ C9- N]tyrosine (m/z 226→179); bromo-[ C6]tyrosine (m/z

13 13 15 260→214); bromo -[ C6]tyrosine (m/z 266→220); bromo -[ C9- N]tyrosine (m/z

12 13 15 270→223); [ C6]tyrosine (m/z 182→136); and [ C9 N1]tyrosine (m/z 192→145). The

108 maximum ion injection time was 100 ms; a scan rate was used that permitted a minimum sampling rate of at least 9 points/chromatographic peak. For all analyses, results were normalized to the content of the precursor amino acid L-tyrosine, which was monitored within the same injection of each oxidized amino acid. The results are listed in Tables 3.6 and 3.7.

Table 3.6. Nitrotyrosine, chlorotyrosine and bromotyrosine levels in autistic patients.

nitroTyr/Tyr ClTyr/Tyr BrTyr/Tyr Patient code (mol/mol) (mol/mol) (mol/mol)

A1 3.7300e-6 1.0700e-6 4.8871e-5

A2 6.9951e-6 5.8656e-6 3.4013e-5

A3 7.5331e-6 8.5004e-6 4.6604e-5

A4 6.4280e-6 1.0165e-5 3.5161e-5

A5 8.6893e-6 6.3993e-6 3.8037e-5

A6 6.2144e-6 6.4514e-6 3.1739e-5

A7 8.7463e-6 1.2837e-5 4.8275e-5

A8 5.8117e-6 9.2790e-6 3.7834e-5

A9 5.4147e-6 7.2821e-6 3.9738e-5

A10 2.0685e-6 1.4240e-5 3.9119e-5

A11 1.2195e-5 7.3088e-6 4.0735e-5

A12 7.3582e-6 6.2857e-6 3.6257e-5

A13 1.0113e-5 3.3894e-5 3.9214e-5

A14 4.8539e-6 2.7447e-5 5.1203e-5

A15 1.0264e-5 4.1406e-5 4.3703e-5

109 A16 1.1858e-5 2.7301e-5 4.7188e-5

A17 8.7110e-6 1.4430e-5 4.3352e-5

A18 1.2641e-5 1.3353e-5 3.9286e-5

A19 3.4437e-6 5.4245e-6 3.9175e-5

A20 1.7499e-5 2.0681e-5 4.7605e-5

A21 6.9135e-6 3.7879e-6 5.5540e-5

A22 9.3421e-6 1.9860e-5 3.6565e-5

A23 1.0140e-5 3.2724e-5 3.7768e-5

A24 5.1831e-6 6.1468e-6 4.1817e-5

A25 6.2409e-6 8.8281e-6 3.9079e-5

A26 6.3528e-6 4.4593e-6 3.9699e-5

A27 6.0950e-6 5.1037e-6 4.7011e-5

Average 7.8087e-6 1.3710e-5 4.1651e-5

STDEV 3.2943e-6 1.0338e-5 5.6909e-6

Table 3.7. Nitrotyrosine, chlorotyrosine and bromotyrosine levels in normal controls.

nitroTyr/Tyr ClTyr/Tyr BrTyr/Tyr Normal code (mol/mol) (mol/mol) (mol/mol)

N1 9.5160e-6 6.8757e-6 3.1430e-5

N2 8.0010e-6 1.0466e-5 3.1475e-5

N3 6.3955e-6 2.5877e-5 4.2179e-5

N4 8.5125e-6 1.7408e-5 3.9366e-5

N5 6.4621e-6 9.1273e-6 4.0811e-5

110 N6 6.0418e-6 2.7017e-6 2.8182e-5

N7 1.1408e-5 2.7262e-5 4.9403e-5

N8 7.2531e-6 3.3149e-5 2.9346e-5

N9 6.3484e-6 3.1250e-6 4.6537e-5

N10 8.4462e-6 2.8620e-5 4.3595e-5

N11 8.2132e-6 5.5500e-6 3.2361e-5

Average 7.87e-6 1.55e-5 3.77e-5

STDEV 1.62e-6 1.14e-5 7.41e-6

111 3.6. References

1. Kidd, P. M., Autism, an extreme challenge to integrative medicine. Part 1: the

knowledge base. Alternative Medicine Review. 2002,

http://www.findarticles.com/p/articles/mi_m0FDN/is_4_7/ai_91155402.

2. Edelson, S. M., Overview of autism. http://www.autism.org/overview.html.

3. Kanner, L., Autistic disturbances of affective contact. Nervous Child 1943, 2, 217-

250.

4. Bryson, S. E.; Smith, I. M., Epidemiology of autism: prevalence, associated

characteristics, and implications for research and service delivery. Ment. Retard Dev.

Disabil. Res. Rev. 1998, 4, 97-103.

5. Persson, B., Brief report: A longitudinal study of quality of life and independence

among adult men with autism. J. Autism Dev. Disord. 2000, 30, 61-66.

6. Korvatska, E.; Van De Water, J.; Anders, T. F.; Gershwin, M. E., Genetic and

immunologic considerations in autism. Neurobiol. Dis. 2002, 9, 107-125.

7. Lord C, C. E., Leventhal BL, Amaral DG. , Autism spectrum disorders. Neuron.

2000, 28, 355-363.

8. Wakefield, A. J.; Murch, S. H.; Anthony, A., et al. , Ileal-lymphoid-nodular hyperplasia, nonspecific colitis, and pervasive developmental disorder in children. Lancet.

1998, 351, 637-641.

9. James, S. J.; Cutler, P.; Melnyk, S.; Hernigan, S.; Janak, L.; Gaylor, D. W.;

Neubrander, J. A., Metabolic biomarkers of increased oxidative stress and methylation

capacity in children with autism. Am. J. Clin. Nutr. 2004, 80, 1611-1617.

112 10. Hviid, A.; Stellfeld, M.; Wohlfahrt, J.; Melbye, M., Association between thimerosal-containing vaccine and autism. JAMA 2003, 290, 1763-1766.

11. Walsh, W. J.; Usman, A.; Tarpey, J., American Psychiatric Association Annual

Meeting; New Orleans, LA. 2001.

12. Deneke, S. M., Thiol-based antioxidants. Curr. Top Cell Regul. 2000, 36, 151-180.

13. Freedman, J. H.; Ciriolo, M. R.; Peisach, J., The role of glutathione in copper metabolism and toxicity. J. Biol. Chem. 1989, 264, 5598-5605.

14. Jiang, J.; St Croix, C. M.; Sussman, N.; Zhao, Q.; Pitt, B. R.; Kagan, V. E.,

Contribution of glutathione and metallothioneins to protection against copper toxicity and redox cycling: quantitative analysis using MT+/+ and MT-/- mouse lung fibroblast cells.

Chem. Res. Toxicol. 2002, 15, 1080-1087.

15. James, S. J.; Cutler, P.; Melnyk, S.; Hernigan, S.; Janak, L.; Gaylor, D. W.;

Neubrander, J. A., Metabolic biomarkers of increased oxidative stress and methylation capacity in children with autism. Am. J. Clin. Nutr. 2004, 80, 1611-1617.

16. Waring, R. H.; Klovrza, L. V., Sulphur Metabolism in Autism. J. Nutri. Enviro.

Med. 2000, 10, 25-32.

17. Golse, B.; Debray-Ritzen, P.; Durosay, P.; Puget, K.; Michelson, A. M., Alterations in two enzymes: superoxide dismutase and glutathion peroxidase in developmental infantile psychosi (infantile autism) (author's transl). Rev. Neurol. (Paris) 1978, 134, 699-

705.

18. Warren, R. P.; Foster, A.; Margaretten, N. C., Immune abnormalities in patients with autism. J. Autism Dev. Disord. 1987, 16, 189-197.

113 19. Gupta, S.; Aggarwal, S.; Heads, C., Dysregulated immune system in children with

autism: Beneficial effects of intravenous immune globulin on autistic characteristics. J.

Autism Dev. Disord. 1996, 26, 439-452.

20. Gupta, S.; Aggarwal, S.; Rashanravan, B.; Lee, T., Th1- and Th2-like cytokines in

CD4+ and CD8+ T cells in autism. J. Neuroimmunol. 1998, 85, (1), 106-9.

21. Connolly, A. M.; Chez, M. G.; Pestronk, A.; Arnold, S. T.; Mehta, S.; Deuel, R. K.,

Serum autoantibodies to brain in Landau-Kleffner variant, autism, and other neurologic disorders. J. Pediatr. 1999, 134, 607-613.

22. Vojdani, A.; Campbell, A. W.; Anyanwu, E.; Kashanian, A.; Bock, K.; Vojdani, E.,

Antibodies to neuron-specific antigens in children with autism: possible cross-reaction

with encephalitogenic proteins from milk, Chlamydia pneumoniae and Streptococcus

group A. J. Neuroimmunol. 2002, 129, (1-2), 168-177.

23. Gu, X.; Meer, S. G.; Miyagi, M.; Rayborn, M. E.; Hollyfield, J. G.; Crabb, J. W.;

Salomon, R. G., Carboxyethylpyrrole protein adducts and autoantibodies, biomarkers for age-related macular degeneration. J. Biol. Chem. 2003, 278, 42027-42035.

24. Evans, T.; Siedlak, S.; Lu, L.; Fu, X.; Wang, Z.; McGinnis, W.; Fakhoury, E.;

Castellani, R.; Hazen, S. L.; Walsh, W. J.; Salomon, R. G.; Smith, M. A.; Perry, G.; Zhu,

X., The Autistic Phenotype Exhibits a Remarkably Localized Modification of Brain

Protein by Products of Free Radical-Induced Lipid Oxidation. Am. J. Biochem.

Biotechnol. 2007, In press.

25. Miyagi, M.; Sakaguchi, H.; Darrow, R. M.; Yan, L.; West, K. A.; Aulak, K. S.;

Stuehr, D. J.; Holleyfield, J. G.; Organisciak, D. T.; Crabb, J. W., Evidence that light modulateds protein nitration in rat retina. Mol. Cell Proteomics. 2002, 1, 293-303.

114 26. Salomon, R. G.; Subbanagounder, G.; Singh, U.; O'Neil, J.; and Hoff, H., Oxidation

of LDL produces levuglandin-protein adducts. Chem. Res. Toxicol. 1997, 10, 750-759.

27. Nicholls, S. J.; Shen, Z.; Fu, X.; Levison, B. S.; Hazen, S. L., Quantification of 3-

nitrotyrosine levels using a benchtop ion trap mass spectrometry method. Methods

Enzymol. 2005, 396, 245-266.

28. Money J.; Bobrow N. A.; C., C. F., Autism and autoimmune disease: a family study.

J. Autism Child Schizophrenia. 1971, 1, 146-160.

29. Comi A. M.; Zimmerman A. W.; Frye V. H.; Law P. A.; N., P. J., Familial

clustering of autoimmune disorders and evaluation of medical risk factors in autism. J.

Child Neurol. 1999, 14, 388-394.

30. Sweeten T. L.; Bowyer S. L.; Posey D. J.; Halberstadt G. M.; J., M. C., Increased

prevalence of familial autoimmunity in probands with pervasive developmental disorders.

Pediatrics. 2003, 112, e420.

31. Jyonouchi H; Sun S; H, L., Proinflammatory and regulatory cytokine production associated with innate and adaptive immune responses in children with autism spectrum disorders and developmental regression. J. Neuroimmunol. 2001, 120, 170-179.

32. Halliwell, B.; Zhao, K.; and Whiteman, M., The ugly, the uglier and the not so good:

a personal view of recent controversies. Free Radic. Res. 1999, 31, 651-669.

33. Smith, P. K.; Krohn, R. I.; Hermanson, G. T.; Mallia, A. K.; Gartner, F. H.;

Provenzano, M. D.; Fujimoto, E. K.; Goeke, N. M.; Olson, B. J.; Klenk, D. C.,

Measurement of Protein Using Bicinchoninic Acid. Anal. Chem. 1985, 150, 76-85.

115 34. DeCaprio, A. P.; Jackowshi, S. J.; and Regan, K. A., Mechanism of formation and quantitation of imines, pyrroles, and stable nonpyrrole adducts in 2,5-hexanedione-treated protein. Mol. Pharmacol. 1987, 32, 542-548.

35. Brennan, M. L.; Wu, W. J.; Fu, X. M.; Shen, Z. Z.; Song, W.; Frost, H.; Vadseth, C.;

Narine, L.; Lenkiewicz, E.; Borchers, M. T.; Lusis, A. J.; Lee, J. J.; Lee, N. A.; Abu-Soud,

H. M.; Ischiropoulos, H.; Hazen, S. L., A tale of two controversies - Defining both the role of peroxidases in nitrotyrosine formation in vivo using eosinophil peroxidase and myeloperoxidase-deficient mice, and the nature of peroxidase-generated reactive nitrogen species. J. Biol. Chem. 2002, 277, (20), 17415-17427.

116

Chapter 4

Serum Vitamin E and Oxidative Protein Modifications in Hemodialysis

117 4.1. Background

In the early 1990’s epidemiologic studies established an inverse relationship between

taking supplemental vitamin E and cardiovascular disease1, 2 or cancer.3 By 2000, an estimated 23 million Americans took supplemental vitamin E.4 However large

prospective randomized controlled trials of treatment failed to show improved

cardiovascular or other health outcomes.5 Thus, proponents of antioxidant therapy argued

that its greatest utility might be found in the setting of abnormal levels of oxidative stress, such as in patients with end stage renal disease (ESRD) treated by dialysis.6

Excess oxidant production and decreased antioxidant defense7 have been recognized

characteristics of the uremic state. In end stage renal disease (ESRD), oral vitamin E

supplementation has been shown to lower the susceptibility of low-density lipoprotein

(LDL) to oxidation,8, 9 and to prevent the oxidative stress associated with intravenous iron

administration for treatment of anemia.10 The SPACE trial showed a significant decrease in cardiovascular events in 196 dialysis patients with pre-existing cardiovascular disease taking oral vitamin E supplements.11 These hopeful results emphasized the importance of

understanding basic mechanisms of action of antioxidants in the uremic milieu.

The clinical observation that vitamin E was protective against morbid events in some

settings,11, 12 but not in all, has been named the antioxidant paradox.6 Factors that lead to

oxidative stress in living systems are complex. For example, the kinetics of reaction

between vitamin E and oxidants are slower than the rate at which free radical species

inactivate nitric oxide.13 Therefore vitamin E would not be expected to correct

118 abnormalities in flow-mediated vasodilation14-16 despite effectively decreasing the

oxidation of LDL.8, 9 In addition, vitamin E has been shown to act as a pro-oxidant, particularly in the absence of .17, 18

Patients with ESRD have increased quantities of oxidatively modified circulating and

tissue proteins – including advanced glycation end products (AGEs),19, 20 advanced

oxidative protein products (AOPP),21, 22 oxidized low density lipoprotein (oxLDL),23

(E)-4-hydroxy-2- nonenal (HNE)-derived-2-pentylpyrroles (PP) and isolevuglandin adducts24, 25 – as well as oxidized lipids including isoprostanes26-29 and oxysterols.30 The goal of this project was to discover the effect of supplemental vitamin E on levels of oxidative protein modifications. We measured pentosidine, a glycoxidation-derived protein modification, as well as protein modifications derived from the lipid peroxidation products, iso[4]levuglandin (iso[4]LGE2), HNE, and (E)-4-oxo-2-nonenal (ONE), in a highly oxidative clinical state – ESRD treated by hemodialysis. We explored the relationships between the levels of these products at baseline and over the course of

treatment, and examined the relationship between levels of oxidation products and factors

known to influence oxidation in the clinical setting.

119 4.2. Results

The demographic characteristics of the 34 study subjects are summarized in Table 4.1.

Table 4.1. Baseline clinical characteristics of patients.

Placebo Group Treatment Group P value ∗

Number of Patients 17 17

Gender (male/female) 8/9 6/11 .48

Race (African American/Caucasian) 13/4 16/1 .13

Diabetes (no/yes) 7/10 6/11 .72

Age (years) 54.7 56.7 .67 ±13.5 ±14.1

Time on dialysis at start of study 47 44 .83 (M) ±49 ±34 Creatinine (mg/dL) 9.9 9.6 .79 ±3.4 ±2.2 Urea (md/dL) 51 44 .24 ±19 ±15

Standardized Kt/V (dimensionless 1.42 1.43 .92 measure of dialysis adequacy) ±.30 ±.34 Body Mass Index (kg/m2) 34.4 30.1 .18 ±9.7 ±8.1

∗ For significant difference in mean values for treatment group versus controls.

Individual participation in the study ranged between 2.1-20.6 months (mean follow up

9.8 ± 4.7). During the course of the study 17 subjects developed thrombotic events

120 involving their hemodialysis vascular accesses, as previously described.31 There were no differences in numbers of events between placebo and vitamin E treatment groups. Nor were there differences in numbers of events between patients with diabetes and without diabetes. During the course of the study 1 patient received a transplant, 1 transferred out of the city, 4 patients had documented myocardial infarctions, and there were 5 deaths.

Clinical laboratory measurements of total iron, iron binding protein and ferritin, were not found to influence levels of glycoxidation or lipid peroxidation products. Nor was there a dose effect related to the quantity of exogenous intravenous iron and erythropoietin administered as part of the routine treatment of anemia in patients on dialysis (data are shown in Appendix Tables S1-S3).

α-Tocopherol levels increased two fold in treated patients (Table 4.2). In contrast

γ-tocopherol levels dropped to half of baseline in patients treated with oral vitamin E alpha (α-tocopherol) (Table 4.2). At baseline there was a positive correlation between levels of α- and γ-tocopherol, Fig 4.1 A. A positive correlation continued in the placebo group. After treatment with vitamin E alpha, the correlation between plasma levels of α- and γ-tocopherol became negative, with the highest levels of α-tocopherol associated with the lowest levels of γ-tocopherol (Table 4.2), and shown at the 3 month follow up time point (Fig 4.1B).

121 Table 4.2. Plasma levels of alpha and gamma tocopherol in placebo and vitamin E treated patients.

Time point Baseline 3 months 6 months 9 months

Alpha Tocopherol Time p

Placebo 12.15 12.44 12.76 13.79 .74

SD 3.48 3.44 4.00 5.31

N 17 14 13 12

Treatment 12.78 26.02 27.27 30.19 .0042

SD 4.52 16.95 14.31 16.40

N 17 16 14 11

Placebo vs Treatment p .65 .006 .0017 .0035

Gamma Tocopherol Time p

Placebo 3.68 3.47 3.51 3.97 .69

SD 2.10 1.69 1.62 2.05

N 17 14 13 12

Treatment 3.92 2.30 1.45 1.49 .0001

SD 1.54 1.47 1.05 1.08

N 17 16 14 11

Placebo vs Treatment p .70 .051 .0006 .0017

122 A B

10 10

8 8

6 6

4 4

2 2

gamma tocopherol (µg/mL) 0 0 0 10 20 30 0 10 20 30 40 50 60 70

alpha tocopherol (µg/mL) Figure 4.1. Relationship between plasma levels of Vitamin E alpha and gamma

before and after treatment. A. Baseline R = 0.46, p = 0.0055. B. The positive

correlation between vitamin E alpha and gamma is present in the placebo group, left

regression line (R = 0.42, p = 0.05). In the patients treated with vitamin E, there is a

negative correlation between the two metabolites, right regression line (R = 0.11,

“not significant”). Gray symbols, placebo. Black symbols, Vitamin E treatment.

Open circles represent baseline; squares, 3 month values.

There were no significant changes in circulating levels of oxidative protein modifications over the course of the study (Table 4.3 and Figures 4.2-4.3). The data set displayed wide standard deviations for pentosidine and ONE-protein adduct. Despite blinded randomization, mean values at baseline were not balanced. In addition an apparent increase in pentosidine and ONE-protein adduct in patients treated by vitamin E alpha was not statistically significant at baseline or at any follow up time point. At baseline, the regression relationships between the lipid peroxidation-derived oxidative

123 protein modifications and both α- and γ-tocopherol were positive, with the highest correlation coefficients for iso[4]levuglandin-protein adducts (Table 4.4).

Table 4.3. Plasma glycoxidation and protein-lipid oxidation products in placebo and vitamin E treated patients.

Time point Baseline 3 months 6 months 9 months pentosidine (pmol/mg protein) Time p Placebo 14.76 15.73 15.65 15.66 .99 SD 9.99 11.18 12.28 15.04 Treatment 18.41 18.93 21.33 23.07 .63 SD 8.65 9.88 9.04 14.01 Placebo vs Treatment p .26 .41 .18 .27 iso[4]LGE2 (nmol/ml) Time p Placebo 8.43 8.84 8.31 8.39 .94 SD 2.68 2.32 2.55 1.69 Treatment 8.26 8.39 8.46 9.90 .29 SD 1.99 2.56 2.37 1.86 Placebo vs Treatment p .85 .64 .87 .06 HNE (nmol/ml) Time p Placebo 0.46 0.49 0.51 0.49 .78 SD 0.13 0.11 0.11 0.12 Treatment 0.48 0.45 0.51 0.51 .43 SD 0.11 0.09 0.08 0.11 Placebo vs Treatment p .72 .37 .89 .63 ONE (pmol/mg protein) Time p Placebo 189 189 189 194 .91 SD 60 65 44 56

124 Treatment 236 204 227 243 .54 SD 68 61 72 102 Placebo vs Treatment p .07 .52 .12 .17

0.55

HNE (nmol/mL) 0.5 placebo 0.45 vitamin E alpha 0.4 treated

0.35

0.3 Baseline 3months 6months 9months

Figure 4.2. Levels of HNE-derived pentylpyrrole over the course of study.

12 Iso[4]LGE 11

10

2 placebo (nmol/mL) 9 vitamin E alpha 8 treatment 7

6

5 Baseline 3months 6months 9months

Figure 4.3. Levels of iso[4]LGE2-protein adduct over the course of study.

Iso[4]-levuglandin-protein adduct level was correlated positively with both α- and

γ-tocopherol levels at baseline, during placebo treatment and during treatment with

125 α-tocopherol (Tables 4.5 and 4.6). HNE-protein adduct level was correlated positively with α-tocopherol at baseline (Table 4.4). In addition at baseline, the correlations among the lipid oxidation products were highly significant (Table 4.7). In contrast, there was no correlation between the glycoxidation product pentosidine and any of the lipid peroxidation protein adducts (Table 4.7), and the regression relationship between the glycoxidation product pentosidine and γ-tocopherol was negative (Table 4.4, p = 0.08).

Table 4.4. Linear regression of glycoxidation and protein-lipid oxidation products with alpha or gamma tocopherol at baseline.

pentosidine HNE iso[4]LGE2 ONE

alpha tocopherol -.02 .46 .42 .29 R .92 .01 .03 .12 p gamma tocopherol -.30 .01 .44 .25 R .08 .95 .02 .20 p

Table 4.5. Linear regression of iso[4]LGE2 with alpha or gamma tocopherol during placebo treatment at 3 months, 6 months and 9 months.

3 months 6 months 9 months alpha tocopherol .1 .07 .2 R .29 .42 .12 p gamma tocopherol .01 .1 .4 R .71 .27 .02 p

126 Table 4.6. Linear regression of iso[4]LGE2 with alpha or gamma tocopherol during

treatment with α-tocopherol at 3 months, 6 months and 9 months.

3 months 6 months 9 months alpha tocopherol .07 .53 .13 R .4 .003 . 3 p gamma tocopherol .03 .006 .25 R .58 .79 .12 p

Table 4.7. Linear regression matrix for glycoxidation and protein-lipid oxidation products at baseline.

pentosidine HNE iso[4]LGE2

HNE .01 R .96 p

iso[4]LGE2 -.05 .39 R .80 .04 p

ONE .04 .41 .37 R .85 .03 .05 p

127 4.3. Discussion

The term “vitamin E” refers to a group of eight naturally occurring chromanols,

including four tocopherols (α, β, γ, δ) and four (α, β, γ, δ). In addition, there

are several stereoisomers of each form.32 Although the naturally occurring RRR-

stereoisomer of α-tocopherol is marketed as “natural vitamin E”, normal dietary

quantities of γ-tocopherol are greater than alpha tocopherol.33 Nevertheless, α-tocopherol

represents the majority of plasma vitamin E.34 This is because α- tocopherol has the

greatest binding affinity to α-tocopherol transfer protein (α-TTP).35 The highly specific

α-TTP preferentially transports α-tocopherol into the blood stream.36 Notably, oral

supplementation with α-tocopherol decreases circulating and tissue levels of γ-tocopherol in normals,37, 38 the elderly,39 and patients with ESRD.28, 32 Presumably, supplementation

with α-tocopherol competitively inhibits α-TPP mediated absorption of dietary

γ-tocopherol.

α-Tocopherol levels are in the normal range in patients with ESRD,32 whereas

γ-tocopherol levels may be normal (as in this study),28 decreased40 or increased.32 Patients in this study received 800 IU of natural RRR-α-tocopherol per day, a dose and form reported to be effective in preventing secondary coronary events in patients with ESRD,11

and in preventing restenosis after percutaneous transluminal coronary angioplasty

(PTCA).41 The power of this study was too small to show an effect of treatment on

cardiovascular outcome. However we have reported previously on an association

128 between lower baseline levels of α- and γ-tocopherol and subsequent thrombotic events

in the hemodialysis (HD) vascular access.31

One important activity of the E is their ability to function as lipid soluble chain-breaking antioxidants in biological membranes. Vitamin E is hypothesized to protect against atherosclerosis through limiting LDL oxidation in the arterial wall.

Unfortunately there is little evidence that vitamin E inhibits LDL oxidation in healthy humans lacking signs of increased oxidative stress.42 Thus, when α-tocopherol is given

for primary prevention, there is no benefit against atherosclerosis or cancer as shown in

clinical trials such as the Gruppo Italiano per lo Studio della Sopravvivenza nell’Infarto

miocardio (GISSI prevention),5 and the Heart Outcomes Prevention Evaluation

(HOPE).43, 44 In secondary prevention, the Cambridge Heart Antioxidant Study (CHAOS)

did show benefit of α-tocopherol supplementation – markedly reducing non-fatal

myocardial infarction in patients with documented coronary artery disease.12 In contrast,

in the HOPE study, there was evidence of harm associated with α-tocopherol

supplementation, with significant increases in heart failure and trends towards increased ischemic complications of atherosclerosis.43, 45, 46

α-Tocopherol is considered the “most potent” non-specific lipid-soluble antioxidant in

the body.47 However, only small differences are expected in abilities of the various

tocopherols and tocotrienols to neutralize free radicals by hydrogen atom transfer. For example, the rate constants for H-atom transfer to a peroxyl radical from α−tocopherol

and γ-tocopherol are 23.5, 15.9 x 105 M-1S-1, respectively.48 Therefore, all of the forms of

129 vitamin E are expected to be similarly effective as chain-breaking antioxidants for inhibiting lipid peroxidation. If the dominant activity of E vitamins was to prevent lipid peroxidation by neutralizing free radicals through hydrogen atom transfer, levels of lipid peroxidation should decrease as those of the E vitamins increase. Two observations of the present study are not in accord with this expectation: (1) there are striking positive correlations between circulating levels of lipid peroxidation products and circulating levels of both α- or γ-tocopherol at baseline and (2) the huge increase in the circulating level of α-tocopherol accomplished by supplementation is not accompanied by a significant decrease in levels of lipid peroxidation products.

The striking positive correlations seen in the present study, between lipid peroxidation products and those of tocopherols at baseline, suggest that in the chronically oxidizing environment of ESRD, the E vitamins are exerting prooxidative effects. Tocopherols may promote lipid autoxidation by recycling catalytic amounts of transition metal ions to low-valent metals which can initiate free radical oxidative chain reactions by inducing

Fenton and Fenton-like reactions, as shown in Figure 4.4. Tocopheryl quinone is produced by oxidation of α-tocopherol with concomitant reduction of transition metal ions (Figure 4.5) that then mediate reductive cleavage of hydroperoxides to alkoxy radicals.49 Thus, tocopherols can promote the generation of alkoxy radicals from lipid hydroperoxides, and thereby initiate free radical chain reactions that generate more lipid hydroperoxides through autoxidation of lipids.50

130 n+ (n+1)+ Fenton Reacton H2O2 + M OH + OH + M

LOOH + M n+ LO + OH + M (n+1)+ Fenton-like Reaction Figure 4.4. Fenton and Fenton-like reactions.

O O O H C H C H C 31 15 M+(n+1) 31 15 M+n 31 15 H OH OH O

α-tocopherol cation radical radical

O O H C H C H2O HO 31 15 M+(n+1) 31 15 M+n O O H O O

C15H31 α radical cation -tocopherylquinone

Figure 4.5. Reduction of transition metal ions by tocopherols.

Another, more subtle, prooxidant effect of supplementation with α-tocopherol may result from the consequent reduction in levels of γ-tocopherol (Figure 4.1).

Myeloperoxidase (MPO), which represents up to 5% of cell protein in inflammatory phagocytes, functions as a major catalyst for lipid peroxidation.51 We previously showed

that MPO promotes the generation of iso[4]LGE2-protein adducts in vivo in a mouse

model of chronic inflammation.52 α-Tocopherol is ineffective against MPO-mediated

52 oxidation. In contrast γ-tocopherol may have unique specificity for protection against

reactive nitrogen species.53

Further complexity arises from other biological activities of vitamin E. In addition to its antioxidant functions, vitamin E is involved in cell signaling, gene expression,

immune response and apoptosis.54 γ-Tocopherol is metabolized largely to

131 2,7,8-trimethyl-2-(β-carboxyethyl)-6-hydroxychroman (γ -CEHC).55 Both γ -tocopherol

and γ-CEHC, but not α-tocopherol, inhibit cyclooxygenase activity and are, thus, anti-inflammatory.56 γ-Tocopherol is more potent than α-tocopherol in modulating

inflammatory pathways.56, 57 Oral supplementation with α-tocopherol results in a

decrease in circulating levels of γ-tocopherol with a resultant increase in inflammation in

unstable atherosclerotic lesions.58

In ESRD chronic inflammation is a major source of increased oxidative stress.7, 59

Untreated hemodialysis membranes activate complement and the immune system during

dialysis.60 In contrast with the present study, dialysis membranes coated with

α-tocopherol decrease the peroxidation of red blood cell and plasma lipids.61 In addition,

hemodialysis with α-tocopherol coated dialysis membranes normalizes endothelial response62 and corrects the over-production of cytokines usually associated with dialysis, normalizing immune function.63, 64 Dialysis with α-tocopherol coated dialyzers decreased

levels of AGEs,65, 66 and of free circulating HNE.67 In the present study, circulating levels

of AGEs, i.e., pentosidine, were inversely related to levels of γ-tocopherol, particularly in

the multivariate model, suggesting that inflammatory pathways that are modulated

normally by γ-tocopherol influence pentosidine formation. Evidence that α-tocopherol

supplementation can increase inflammation is demonstrated in hemodialysis patients who took a 14 day course of this vitamin. Interleukin-6 levels increased in response to treatment, but c-reactive protein levels decreased when the patients received supplements rich in γ-tocopherol.32 Our data emphasize an important contrast between the action of

132 α-tocopherol localized to the site of oxidative and inflammatory activation on the

hemodialysis membrane, versus the dietary interaction of α-tocopherol as an oral

supplement.58

4.4. Conclusions

In summary, in the clinical setting of oxidative stress experienced by patients with

ESRD treated by hemodialysis, the balance of oxidant/antioxidant systems is highly

complex. Both chemical pathways and inflammatory components appear to be involved

in interactions that lead to the formation of glycoxidation and protein-bound lipid

oxidation products. These data demonstrate that intervention by oral supplementation

with the commercially prevalent “natural vitamin E”, i.e., α-tocopherol, does not result in predictable antioxidant benefit.

133 4.5. Experimental Procedures

Methods

Patients

Thirty-four stable patients treated by HD at one of the Centers for Dialysis Care,

Cleveland were recruited for the study. Informed consent was obtained in compliance

with the Institutional Review Board of University Hospitals of Cleveland. Some patients left the study due to death, transplantation, or the initiation of anticoagulant therapy.

Study Design

Patients were provided with Vitamin E (α-tocopherol, 800 IU) or identical placebo capsules to be taken daily. Investigators and patients were blinded as to treatment.

“Natural vitamin E” (RRR-alpha-tocopherol also known as d-alpha-tocopherol) was the kind gift of Cognis Inc (LaGrange, IL). Clinical data was collected by interview, and chart review. Clinical parameters and routine clinical chemistry and hematology results used in standard care and management were obtained in a single reference clinical laboratory. Results were recorded by chart review every three months throughout the study. The total administered dosages of erythropoietin and intravenous iron were recorded for each 3 month time period (Appendix Tables S1-S3), as were significant clinical events, including hospitalizations and surgical interventions.

Assays

At each time point blood was collected in EDTA-metal free tubes for plasma or plain glass tubes for serum. Samples for plasma were placed on ice, and spun in the cold at

134 3000 rpm for 15 minutes. Serum was allowed to clot before centrifugation. Plasma for

analysis of alpha and gamma tocopherol was protected from light at each step. Plasma for

enzyme-linked immunosorbent assay (ELISA) was stabilized prior to storage by addition

of butylated hydroxytoluene (BHT, 10 mM final concentration) and a protease inhibitor

cocktail (Sigma-Aldrich, Milwaukee, WI, Cat. Sigma P8340) (10 μL/mL plasma), as

described.24 After processing, serum and plasma aliquots were quench-frozen in liquid

nitrogen and transported to the laboratory where they were layered with argon and stored

at -80 °C.

Tocopherols were measured by Penny Erhard using a modification of the method of

Sommerburg.68 Briefly, in plasma (200 μL) was added ethanol (200 μL, 200 proof,

Pharmacia) containing alpha tocopherol acetate (32 μg/mL). Hexane (500 μL) was added and the system was allowed to sit in the dark for 5 minutes, vortexed, and then centrifuged at 2000 RPM for 5 mins at 15 °C. The upper hexane layer was saved and the extraction was repeated twice. The hexane extract was dried under nitrogen and reconstituted in methanol. Then the sample (80 μL) was injected into an HPLC system with a Waters UV detector at 292 nm using a reverse phase C18 μBondapack column (3 micron, 15 cm, Waters). The sample was eluted using an isocratic elution with 95% methanol and 5% water at a flow rate of 1 mL/min for 20 mins. Standards for α- and

γ-tocopherol were purchased from Sigma, and prepared freshly for each assay. The intra-assay coefficient of variation was 0.039 for α-tocopherol and 0.041 for γ-tocopherol, and the inter-assay coefficients were 0.078 for both α- and γ-tocopherol . The normal

135 range for α-tocopherol was 12.50 ± 0.23 µg/ml; the normal range for γ-tocopherol was

2.41 ± 0.26 µg/ml. α-Tocopherol levels in plasma from HD patients treated with vitamin

E and placebo at baseline, three months, six months, nine months and twelve months are listed in tables 4.8 and 4.9 respectively. γ-Tocopherol levels in plasma from HD patients treated with vitamin E and placebo at baseline, three months, six months, nine months and twelve months are listed in tables 4.10 and 4.11 respectively.

Table 4.8. α-Tocopherol levels (μg/mL) in plasma from patients treated with vitamin E. Patient Code Baseline 3 months 6 months 9 months 12 months T1 16.04 23.18 56.63 60.81 55.49 T2 15.21 66.53 40.28 T3 8.69 15.97 14.95 14.91 13.24 T4 12.15 17.95 18.95 T5 7.02 15.62 12.69 16.60 14.94 T6 10.93 16.67 28.31 31.06 30.21 T7 14.36 9.19 15.49 16.76 20.55 T8 6.99 61.88 42.63 43.68 37.57 T9 17.65 32.26 25.49 49.29 33.13 T10 15.89 28.02 15.39 13.39 T11 17.80 42.87 48.32 42.26 T12 16.69 14.62 28.46 17.70 T13 12.23 17.83 20.66 25.61 T14 8.46 15.69 13.49 T15 13.94 Average 12.78 26.02 27.27 30.19 29.30 STDEV 4.52 16.95 14.31 16.40 14.78

136 Table 4.9. α-Tocopherol levels (μg/mL) in plasma from HD patients treated with placebo

at baseline, three months, six months, nine months and twelve months.

Patient Code Baseline 3 months 6 months 9 months 12 months P1 16.94 16.80 16.22 18.97 18.17 P2 15.27 16.71 11.81 13.91 12.44 P3 13.50 13.86 14.82 23.87 P4 14.54 15.18 11.54 11.61 15.36 P5 8.95 9.07 7.70 7.70 8.25 P6 20.16 15.52 21.16 19.46 19.01 P7 11.14 12.19 12.77 13.30 13.76 P8 13.34 15.79 17.58 15.76 22.34 P9 10.43 8.92 9.04 10.18 8.68 P10 14.94 13.79 13.56 15.14 P11 11.47 11.56 12.76 10.71 P12 6.43 6.63 7.51 P13 7.66 10.37 9.36 Average 12.15 12.44 12.76 13.79 16.14 STDEV 3.48 3.44 4.00 5.31 7.15

Table 4.10. γ-Tocopherol levels (μg/mL) in plasma from patients treated with vitamin E at baseline, three months, six months, nine months and twelve months.

Patient Code Baseline 3 months 6 months 9 months 12 months T1 6.04 4.98 1.51 2.14 T2 6.19 2.56 0.91 T3 3.49 2.81 2.79 2.72 T4 2.96 0.66 0.41 T5 2.58 4.71 4.16 3.93 1.27

137 T6 3.88 3.67 1.25 0.72 5.41 T7 4.16 1.17 1.23 1.39 T8 2.19 1.15 0.97 0.69 1.51 T9 5.14 2.37 2.19 0.63 2.51 T10 4.35 1.65 2.01 1.72 T11 6.95 0.99 0.77 1.05 T12 5.02 2.71 0.47 0.96 T13 2.69 0.61 0.36 0.39 T14 2.74 0.89 1.24 T15 2.49 Average 3.92 2.30 1.45 1.49 2.68 STDEV 1.54 1.47 1.05 1.08 1.90

Table 4.11. γ-Tocopherol levels (μg/mL) in plasma from HD patients treated with placebo at baseline, three months, six months, nine months and twelve months.

Patient Code Baseline 3 months 6 months 9 months 12 months P1 6.91 5.73 5.25 8.40 2.05 P2 4.38 6.20 3.62 4.23 P3 0.61 0.75 1.38 1.39 2.00 P4 3.01 2.23 2.68 2.85 3.07 P5 5.82 5.65 5.42 4.76 4.72 P6 9.12 4.45 6.46 6.61 P7 2.69 2.90 3.16 2.43 5.20 P8 4.08 5.21 4.91 5.49 0.76 P9 2.08 1.61 1.37 2.98 P10 2.56 2.47 3.64 3.33 P11 2.17 3.34 2.52 3.37

138 P12 3.27 2.66 3.49 P13 2.04 2.94 1.69 Average 3.68 3.47 3.51 3.97 2.97 STDEV 2.10 1.69 1.62 2.05 1.72

Pentosidine was measured by HPLC using a modification of the method of Odetti as

previously published.69, 70 Plasma (500 μL) was subjected to precipitation with 10% trichloroacetic acid (TCA) (500 μL) on ice followed by centrifugation. The pellets were washed twice with 5% cold TCA and acid hydrolyzed in 6 N HCl (1 mL) for 16 h at 110

°C in borosilicate tubes with Teflon coated screw caps. Acid was then removed by vacuum centrifugation. The hydrolyzed pellet was dissolved in 250 μL of water/0.01 M heptafluorobutyric acid. The equivalent of 0.8 mg of plasma protein was injected onto an

HPLC system (Waters). A Vydac type 218 TP C18 column (25 × 0.46 cm, 10 micron)

(Separations Group, Hesperia, CA) was used. The HPLC was programmed with a linear gradient from 1 to 35 min of 10-17% acetonitrile in water and 0.1% heptafluorobutyric acid as a counter ion. Pentosidine eluted at 30 min as monitored by fluorescence excitation at 335 nm and emission at 385 nm. Pentosidine prepared according to a published procedure71 was used as a standard. Results were calculated per milligram

added protein. This work was done by Penny Erhard. The results are listed in tables 4.12

and 4.13 respectively. In previous studies the range for pentosidine in healthy control

subjects was 1.35 ± 0.6 pmol/mg protein; the usual range in patients on HD was 22.9 ±

10.8 pmol/mg protein.72, 73

139 Table 4.12. Pentosidine levels (pmol/mg protein) in plasma from HD patients treated with vitamin E supplementation at baseline, three months, six months, nine months and twelve months.

Patient Code Baseline 3 months 6 months 9 months 12 months

T1 14.68 19.78 31.18 37.20 27.19

T2 18.42 16.90 19.01

T3 28.62 35.41 35.60 43.40 40.43

T4 37.71 18.38 17.36

T5 20.61 18.04 17.50 20.57 23.00

T6 21.92 13.60 17.21 15.30 18.08

T7 5.37 17.27 13.06 19.08 19.85

T8 13.82 9.12 9.02 9.17 9.48

T9 8.36 13.40 13.31 11.52 11.94

T10 10.31 31.36 30.95 22.74

T11 22.91 7.52 8.93 6.80

T12 10.98 37.68 32.54 44.96

T13 26.78 24.58 25.73

T14 19.32 26.47 27.17

T15 28.06

Average 18.41 18.93 21.33 23.07 21.42

STDEV 8.65 9.88 9.04 14.01 10.35

140 Table 4.13. Pentosidine levels (pmol/mg protein) in plasma from HD patients treated with placebo at baseline, three months, six months, nine months and twelve months.

Patient Code Baseline 3 months 6 months 9 months 12 months

P1 10.53 13.80 13.15 13.48 12.11

P2 8.78 7.55 6.73 7.46 6.85

P3 48.09 50.70 52.04 56.82

P4 16.26 15.63 12.13 11.87 10.91

P5 13.62 16.06 15.19 11.36 11.57

P6 8.41 8.55 8.59 9.79 11.07

P7 4.36 5.05 4.91 5.13 6.20

P8 21.91 16.42 17.90 17.86 20.16

P9 5.85 8.69 5.37 6.39

P10 13.82 17.36 16.39 16.46

P11 23.69 23.58 23.10

P12 8.97 9.16 8.94

P13 12.04 13.03 18.99

Average 14.76 15.73 15.65 15.66 11.27

STDEV 9.99 11.18 12.28 15.04 4.57

141 Protein adducts of iso[4]LGE2, HNE derived pentylpyrrole and ONE were measured

by competitive ELISA as described above (Chapter 2). Iso[4]LGE2-BSA and iso[4]LGE2-HSA were synthesized as described in Chapter 3. ON-BSA and ON-HSA

were synthesized as described previously24, and specific antibodies were produced,

purified and characterized.74 For pentylpyrrole immunoreactivity, ON-BSA (final

concentration was 187 pmol/mL) was used as a coating agent. ON-HSA was used as a standard. The initial concentration of ON-HSA was 13800 pmol/mL. A dilution factor of

0.3 was employed for standard and samples. The normal range for HNE–protein adduct was 436 ± 77 pmol/mL; the usual range in patients on HD was 543 ± 190 pmol/mL. The intra-assay and inter-assay coefficient of variation were 5% and 6% respectively. The pentylpyrrole immunoreactivities in plasma from patients treated with vitamin E and placebo were listed in tables 4.14 and 4.15 respectively. For iso[4]LGE2 immunoreactivity, iso[4]LGE2-BSA (final concentration was 22.8 nmol/mL) was used as a coating agent and iso[4]LGE2-HSA was used as a standard. The initial concentration of

iso[4]LGE2-HSA was 810 nmol/mL. A dilution factor of 0.2 was employed for standard and samples. Eight serial dilutions for standard, five serial dilutions of samples were

performed. The normal range for iso[4]LGE2-protein adduct was 4087 ± 47.9 pmol/mL;

the usual range in patients on HD was 8344 ± 2296 pmol/mL.74 The intra-assay and

inter-assay coefficient of variation for iso[4]LGE2-protein adduct were 4.65% and 7.11%

respectively. Iso[4]LGE2 immunoreactivities in plasma from patients treated with vitamin

E and placebo are listed in tables 4.16 and 4.17. ONE -BSA and anti-ONE-RNase

142 antibody were kind gifts of Dr. Lawrence Sayre. ONE-BSA was used as a coating agent

(final concentration was 1170 pmol/mg) and a standard (initial concentration was 11700

pmol/mg). The normal range for ONE-RNase was 114.72 ± 28.73 pmol/mg BSA; the

usual range in patients on HD was 152.58 ± 33.68 pmol/mg BSA. The intra-assay coefficient of variation was 3.36%, and the inter-assay coefficient was 6.92%.

ONE-RNase immunoreactivities in plasma from patients treated with vitamin E and placebo are listed in tables 4.18 and 4.19 respectively.

Statistical analyses

Treatment and placebo groups were compared with respect to demographic and baseline characteristics using paired comparisons for parametric data, and chi square analysis for

descriptive variables. Longitudinal effects of treatment were compared using analysis of

variance (ANOVA) with subsequent pair-wise analysis using a 2-tailed t test. The

significance of group comparisons was corrected for the effect of repeated measures.

Correlations between measured parameters were ascertained using pair-wise comparisons

by linear regression. Models of interactions were constructed using multiple regression analysis and the least squares model of fit. Data were expressed as mean ± SD. Statistical significance was defined as P less than 0.05. All analyses were performed using JMP

5.12 (SAS Institute, Cary NC).

143 Table 4.14. HNE-derived pentylpyrrole immunoreactivities in plasma from patients treated with vitamin E at baseline, 3 months, 6 months, 9 months and 12 months.

Patient Code Baseline 3 months 6 months 9 months 12 months

T1 0.47 0.45 0.46 0.43 0.38

T2 0.53 0.54 0.42

T3 0.50 0.23 0.55 0.49 0.49

T4 0.39 0.43 0.40

T5 0.47 0.41 0.46 0.47 0.47

T6 0.60 0.39 0.70 0.74 0.80

T7 0.50 0.55 0.53 0.56 0.53

T8 0.55 0.55 0.55 0.56 0.58

T9 0.45 0.43 0.50 0.47 0.49

T10 0.56 0.46 0.52 0.44

T11 0.72 0.47 0.61 0.67

T12 0.51 0.61 0.54 0.50

T13 0.38 0.34 0.38 0.35

T14 0.30 0.44 0.47

Average 0.48 0.45 0.51 0.51 0.53

STDEV 0.11 0.09 0.08 0.11 0.13

144 Table 4.15. HNE-derived pentylpyrrole immunoreactivities (nmol/mL) in plasma from patients treated with placebo at baseline, 3 months, 6 months, 9 months and 12 months.

Patient Code Baseline 3 months 6 months 9 months 12 months

P1 0.47 0.40 0.44 0.39 0.43

P2 0.53 0.50 0.61 0.65 0.75

P3 0.53 0.47 0.59 0.60

P4 0.66 0.62 0.67 0.66 0.73

P5 0.30 0.37 0.36 0.29 0.38

P6 0.54 0.51 0.55 0.53 0.58

P7 0.54 0.53 0.56 0.54 0.51

P8 0.22 0.39 0.42 0.42 0.32

P9 0.54 0.66

P10 0.30 0.32 0.36 0.38 0.23

P11 0.49 0.58 0.61 0.39

P12 0.51 0.56 0.55 0.52

P13 0.57 0.55 0.54 0.56

P14 0.31 0.37 0.37

Average 0.46 0.49 0.51 0.49 0.49

STDEV 0.13 0.11 0.11 0.12 0.19

145 Table 4.16. Iso[4]LGE2-protein immunoreactivities (nmol/mL) in plasma from patients treated with vitamin E at baseline, 3 months, 6 months, 9 months and 12 months.

Patient Code Baseline 3 months 6 months 9 months 12 months

T1 12.96 12.21 15.51 13.61 11.71

T2 9.13 9.32 8.22

T3 8.74 4.46 9.60 8.50 9.96

T4 9.29 8.86 7.68

T5 10.10 9.78 8.50 12.00 11.85

T6 9.54 2.85 9.04 11.17 12.56

T7 8.00 8.49 6.60 8.90 8.83

T8 7.37 9.38 9.22 8.76 8.79

T9 8.33 8.32 7.54 8.85 8.86

T10 7.74 6.88 5.66

T11 8.72 8.69 9.19 9.89

T12 7.50 9.79 8.79 9.92

T13 6.09 6.38 6.46 7.43

T14 6.22 7.88 6.52

Average 8.26 8.39 8.46 9.90 11.71

STDEV 1.99 2.56 2.37 1.86 1.64

146 Table 4.17. Iso[4]LGE2-protein immunoreactivities (nmol/mL) in plasma from patients treated with placebo at baseline, 3 months, 6 months, 9 months and 12 months.

Patient Code Baseline 3 months 6 months 9 months 12 months

P1 16.26 15.31 14.94 12.88 14.35

P2 8.46 7.51 8.10 6.68 8.00

P3 7.51 9.03 8.28 8.06

P4 8.79 7.70 8.40 7.74 8.36

P5 7.20 6.93 6.84 6.86 9.70

P6 9.91 8.61 7.64 10.06 8.94

P7 6.36 7.42 8.07 7.72 6.11

P8 5.34 7.14 8.97 7.69 7.43

P9 5.98 8.03 7.62 8.88 4.15

P10 8.61 11.60 8.84 8.73

P11 8.44 9.50 7.73 7.01

P12 8.85 8.85 10.81 8.11

P13 7.92 7.29 5.81

P14

Average 8.43 8.84 8.31 8.39 8.38

STDEV 2.68 2.32 2.55 1.69 2.97

147 Table 4.18. ONE-protein immunoreactivities (pmol/mg protein) in plasma from patients treated with vitamin E at baseline, 3 months, 6 months and 9 months.

Patient Code Baseline 3 months 6 months 9 months

T1 160.5 187.5 118.7 84.6

T2 242.6 234.9 210.4

T3 360.9 151.6 N/A 341.7

T4 244.2 194.1 184.3

T5 252.1 268.9 315.6 437.9

T6 252.1 110.3 280.2 252.1

T7 216.4 211.6 298.2 231.6

T8 221.1 212.8 211.2 208.0

T9 302.5 297.5 335.5 307.7

T10 266.9 202.3 216.4 158.1

T11 339.7 325.5 305.8 325.0

T12 159.7 176.6 143.4 156.9

T13 158.3 128.8 144.7 175.0

T14 130.0 165.7 193.8

Average 236.2 204.9 227.5 243.5

STDEV 68.9 61.1 72.3 102.2

148 Table 4.19. ONE-protein immunoreactivities (pmol/mg protein) in plasma from patients treated with placebo at baseline, 3 months, 6 months and 9 months.

Patient Code Baseline 3 months 6 months 9 months

P1 299.5 224.0 208.8 171.7

P2 168.0 133.7 150.6 165.0

P3 225.8 180.3 203.0 252.1

P4 100.3 92.2 130.1 90.4

P5 151.6 176.4 171.3 146.3

P6 238.5 220.7 236.6 200.0

P7 186.7 157.7 157.7 169.9

P8 99.3 207.5 236.9 276.6

P9 222.4 280.2

P10 223.0 335.5 242.9 280.7

P11 266.9 219.7 202.3 233.6

P12 143.4 134.7 135.9 176.6

P13 192.0 167.2 246.2 175.0

P14 132.3 123.2 138.3

Average 189.3 189.5 189.3 194.8

STDEV 60.4 64.9 43.9 56.3

149 4.6. References

1. Rimm, E. B.; Stampfer, M. J.; Ascherio, A.; Giovannucci, E.; Colditz, G. A.; Willett,

W. C., Vitamin E consumption and the risk of coronary heart disease in men. N. Engl. J.

Med. 1993, 328, (20), 1450-6.

2. Stampfer, M. J.; Hennekens, C. H.; Manson, J. E.; Colditz, G. A.; Rosner, B.; Willett,

W. C., Vitamin E consumption and the risk of coronary disease in women. N. Engl. J.

Med. 1993, 328, (20), 1444-9.

3. Hunter, D. J.; Manson, J. E.; Colditz, G. A.; Stampfer, M. J.; Rosner, B.; Hennekens,

C. H.; Speizer, F. E.; Willett, W. C., A prospective study of the intake of vitamins C, E, and A and the risk of breast cancer. N. Engl. J. Med. 1993, 329, (4), 234-40.

4. Ford, E. S.; Ajani, U. A.; Mokdad, A. H., Brief communication: The prevalence of high intake of vitamin E from the use of supplements among U.S. adults. Ann. Intern.

Med. 2005, 143, (2), 116-20.

5. Dietary supplementation with n-3 polyunsaturated fatty acids and vitamin E after myocardial infarction: results of the GISSI-Prevenzione trial. Gruppo Italiano per lo

Studio della Sopravvivenza nell'Infarto miocardico. Lancet. 1999, 354, (9177), 447-55.

6. Halliwell, B., The antioxidant paradox. Lancet. 2000, 355, 1179-80.

7. Himmelfarb, J.; Stenvinkel, P.; Ikizler, T. A.; Hakim, R. M., The elephant in uremia: oxidant stress as a unifying concept of cardiovascular disease in uremia. Kidney Int. 2002,

62, (5), 1524-38.

150 8. Islam, K. N.; O'Byrne, D.; Devaraj, S.; Palmer, B.; Grundy, S. M.; Jialal, I.,

Alpha-tocopherol supplementation decreases the oxidative susceptibility of LDL in renal failure patients on dialysis therapy. Atherosclerosis 2000, 150, (1), 217-24.

9. Diepeveen, S. H.; Verhoeven, G. W.; Van Der Palen, J.; Dikkeschei, L. D.; Van Tits,

L. J.; Kolsters, G.; Offerman, J. J.; Bilo, H. J.; Stalenhoef, A. F., Effects of atorvastatin and vitamin E on lipoproteins and oxidative stress in dialysis patients: a randomised-controlled trial. J. Intern. Med. 2005, 257, (5), 438-45.

10. Roob, J. M.; Khoschsorur, G.; Tiran, A.; Horina, J. H.; Holzer, H.; Winklhofer-Roob,

B., Vitamin E attenuates oxidative stress induced by intravenous iron in patients on hemodialysis. J. Am. Soc. Nephrol. 2000, 11, 539-549.

11. Boaz, M.; Smetana, S.; Weinstein, T.; Matas, Z.; Gafter, U.; Iaina, A.; Knecht, A.;

Weissgarten, Y.; Brunner, D.; Fainaru, M.; Green, M., Secondary prevention with antioxidants of cardiovascular disease in endstage renal disease (SPACE): randomised placebo-controlled trial. Lancet. 2000, 356, 1213-1218.

12. Stephens, N.; Parsons, A.; Shofield, P.; Kelly, F.; Cheeseman, K.; Mitchinson, M.;

Brown, M., Randomised controlled trial of vitamin E in patients with coronary disease:

Cambridge Heart Antioxidant Study (CHAOS). Lancet. 1996, 347, 781-786.

13. Landmesser, U.; Harrison, D. G.; Drexler, H., Oxidant stress-a major cause of reduced endothelial nitric oxide availability in cardiovascular disease. Eur. J. Clin.

Pharmacol. 2006, 62 Suppl 13, 13-9.

14. Economides, P. A.; Khaodhiar, L.; Caselli, A.; Caballero, A. E.; Keenan, H.; Bursell,

S. E.; King, G. L.; Johnstone, M. T.; Horton, E. S.; Veves, A., The effect of vitamin E on

151 endothelial function of micro- and macrocirculation and left ventricular function in type 1

and type 2 diabetic patients. Diabetes 2005, 54, (1), 204-11.

15. Ghiadoni, L.; Cupisti, A.; Huang, Y.; Mattei, P.; Cardinal, H.; Favilla, S.; Rindi, P.;

Barsotti, G.; Taddei, S.; Salvetti, A., Endothelial dysfunction and oxidative stress in

chronic renal failure. J. Nephrol. 2004, 17, (4), 512-9.

16. Simons, L. A.; von Konigsmark, M.; Simons, J.; Stocker, R.; Celermajer, D. S.,

Vitamin E ingestion does not improve arterial endothelial dysfunction in older adults.

Atherosclerosis 1999, 143, (1), 193-9.

17. Buettner, G., The pecking order of free radicals and antioxidants: lipid peroxidation,

alpha-tocopherol and ascorbate. Arch. Biochem. Biophys. 1993, 300, 535-543.

18. Witting, P. K.; Upston, J. M.; Stocker, R., The molecular action of alpha-tocopherol in lipoprotein lipid peroxidation. Pro- and antioxidant activity of vitamin E in complex heterogeneous lipid emulsions. Subcell Biochem. 1998, 30, 345-90.

19. Weiss, M.; Erhard, P.; Kader-Attia, F.; Wu, Y.; DeOreo, P.; Araki, A.; Monnier, V.,

Mechanisms for the formation of advanced glycation end products (AGEs) in end stage renal disease (ESRD). Kidney Int. 2000, 57, 2571-2585.

20. Agalou, S.; Ahmed, N.; Babaei-Jadidi, R.; Dawnay, A.; Thornalley, P., Profound mishandling of protein glycation degradation products in uremia and dialysis. J. Am. Soc.

Nephrol. 2005, 16, (5), 1471-1485.

21. Witko-Sarsat, V.; Friedlander, M.; Capeillere-Blandin, C.; Nguyen-Khoa, T.;

Nguyen, A.; Zingraff, J.; Jungers, P.; Descamps-Latscha, B., Advanced oxidative protein

152 products as a novel marker of oxidative stress in uremia. Kidney Int. 1996, 49,

1304-1313.

22. Galli, F.; Benedetti, S.; Floridi, A.; Canestrari, F.; Piroddi, M.; Buoncristiani, E.;

Buoncristiani, U., Glycoxidation and inflammatory markers in patients on treatment with

PMMA-based protein-leaking dialyzers. Kidney Int. 2005, 67, (2), 750-9.

23. Maggi, E.; Bellazzi, R.; Falaschi, F.; Frattoni, A.; Perani, G.; Finardi, G.; Gazo, A.;

Nai, M.; Romanini, D.; Bellomo, G., Enhanced LDL oxidation in uremic patients: an additional mechanism for accelerated atherosclerosis? Kidney Int. 1994, 45, 876-883.

24. Salomon, R.; Kaur, K.; Podrez, E.; Hoff, H.; Krushinsky, A.; Sayre, L., HNE-derived

2-pentylpyrroles are generated during oxidation of LDL, are more prevalent in blood plasma from patients with renal disease or atherosclerosis, and are present in atherosclerotic plaques. Chem. Res. Toxicol. 2000, 13, (7), 557-64.

25. Salomon, R.; Kaur, K.; Batyreva, E., Isolevuglandin-protein adducts in oxidized low

density lipoprotein and human plasma: a strong connection with cardiovascular disease.

Trends Cardiovasc. Med. 2000, 10, (2), 53-9.

26. Handelman, G.; Walter, M.; Adhikarla, R.; Gross, J.; Dallai, G.; Lewin, N., Elevated plasma F2-isoprostanes in patients on long-term hemodialysis. Kidney Int. 2001, 59,

1960-1966.

27. Ikizler, T.; Moroz, J.; Roberts, L.; Evanson, J.; Becker, B.; Hakim, R.; Shire, Y.;

Himmelfarb, J., Plasma F2-isprostanes levels are elevated in chronic hemodialysis

patients. Clin. Nephrol. 2002, 58, 190-197.

153 28. Smith, K. S.; Lee, C. L.; Ridlington, J. W.; Leonard, S. W.; Devaraj, S.; Traber, M.

G., Vitamin E supplementation increases circulating vitamin E metabolites tenfold in end-stage renal disease patients. Lipids 2003, 38, (8), 813-9.

29. Wiswedel, I.; Hirsch, D.; Carluccio, F.; Hampl, H.; Siems, W., F2-isoprostanes as biomarkers of lipid peroxidation in patients with chronic renal failure. Biofactors 2005,

24, (1-4), 201-8.

30. Siems, W.; Quast, S.; Peter, D.; Augustin, W.; Carluccio, F.; Grune, T.; Sevanian, A.;

Hampl, H.; Wiswedel, I., Oxysterols are increased in plasma of end-stage renal disease patients. Kidney Blood Press. Res. 2005, 28, (5-6), 302-6.

31. Schwing, W.; Erhard, P.; Hollamon, C.; Weigel, K.; Blankschaen, S.; Anderson, J.;

Siegal, C.; Seaman, D.; Valente, J.; DeOreo, P.; Weiss, M., Thrombotic events and markers of oxidation and inflammation in hemodialysis. Hemodialysis Int. 2004, 8,

338-343.

32. Himmelfarb, J.; Kane, J.; McMonagle, E.; Zaltas, E.; Bobzin, S.; Boddupalli, S.;

Phinney, S.; Miller, G., Alpha and gamma tocopherol metabolism in healthy subjects and patients with end-stage renal disease. Kidney Intern. 2003, 64, 978-991.

33. Jiang, Q.; Christen, S.; Shigenaga, M. K.; Ames, B. N., gamma-tocopherol, the major form of vitamin E in the US diet, deserves more attention. Am. J. Clin. Nutr. 2001, 74, (6),

714-22.

34. Winklhofer-Roob, B. M.; van't Hof, M. A.; Shmerling, D. H., Reference values for plasma concentrations of vitamin E and A and carotenoids in a Swiss population from infancy to adulthood, adjusted for seasonal influences. Clin. Chem. 1997, 43, (1), 146-53.

154 35. Traber, M. G.; Arai, H., Molecular mechanisms of vitamin E transport. Annu. Rev.

Nutr. 1999, 19, 343-355.

36. Hosomi, A.; Arita, M.; Sato, Y.; Kiyose, C.; Ueda, T.; Igarashi, O.; Arai, H.; Inoue,

K., Affinity for alpha-tocopherol transfer protein as a determinant of the biological

activities of vitamin E analogs. FEBS Lett. 1997, 409, (1), 105-8.

37. Handelman, G. J.; Machlin, L. J.; Fitch, K.; Weiter, J. J.; Dratz, E. A., Oral

alpha-tocopherol supplements decrease plasma gamma-tocopherol levels in humans. J.

Nutr. 1985, 115, (6), 807-13.

38. Handelman, G. J.; Epstein, W. L.; Peerson, J.; Spiegelman, D.; Machlin, L. J.; Dratz,

E. A., Human adipose alpha-tocopherol and gamma-tocopherol kinetics during and after

1 y of alpha-tocopherol supplementation. Am. J. Clin. Nutr. 1994, 59, (5), 1025-32.

39. Winklhofer-Roob, B. M.; Meinitzer, A.; Maritschnegg, M.; Roob, J. M.;

Khoschsorur, G.; Ribalta, J.; Sundl, I.; Wuga, S.; Wonisch, W.; Tiran, B.; Rock, E.,

Effects of vitamin E depletion/repletion on biomarkers of oxidative stress in healthy aging. Ann. N. Y. Acad. Sci. 2004, 1031, 361-4.

40. Galli, F.; Buoncristiani, U.; Conte, C.; Aisa, C.; Floridi, A., Vitamin E in uremia and dialysis patients. Ann. N.Y. Acad. Sci. 2004, 1031, 348-351.

41. DeMaio, S.; King, S.; Lembo, N.; Roubin, G.; Hearn, J.; Bhagavan, H.; Sgoutas, D.,

Vitamin E supplementation, plasma lipids and incidence of restenosis after percutaneous transluminal coronary angioplasty (PTCA). J. Am. Coll. Nutr. 1992, 11, 68-73.

42. Meagher, E.; Rader, D. J., Antioxidant therapy and atherosclerosis: animal and human studies. Trends Cardiovasc. Med. 2001, 11, (3-4), 162-5.

155 43. Lonn, E.; Bosch, J.; Yusuf, S.; Sheridan, P.; Pogue, J.; Arnold, J. M.; Ross, C.;

Arnold, A.; Sleight, P.; Probstfield, J.; Dagenais, G. R., Effects of long-term vitamin E supplementation on cardiovascular events and cancer: a randomized controlled trial.

JAMA 2005, 293, (11), 1338-47.

44. Brown, B. G.; Crowley, J., Is there any hope for vitamin E? JAMA 2005, 293, (11),

1387-90.

45. Brown, B. G.; Zhao, X. Q.; Chait, A.; Fisher, L. D.; Cheung, M. C.; Morse, J. S.;

Dowdy, A. A.; Marino, E. K.; Bolson, E. L.; Alaupovic, P.; Frohlich, J.; Albers, J. J.,

Simvastatin and niacin, antioxidant vitamins, or the combination for the prevention of coronary disease. N. Engl. J. Med. 2001, 345, (22), 1583-92.

46. MRC/BHF Heart protection study of antioxidant vitamin supplementation in 20,536 high-risk individuals: a randomised placebo-controlled trial. Lancet. 2002, 360, (9326),

23-33.

47. Saldeen, T.; Li, D.; Mehta, J. L., Differential effects of alpha- and gamma-tocopherol on low-density lipoprotein oxidation, superoxide activity, platelet aggregation and arterial thrombogenesis. J. Am. Coll. Cardiol. 1999, 34, (4), 1208-15.

48. Burton, G. W. a. I., K. U., Autoxidation of biological molecules. 1. The antioxidant activity of vitamin e and related chain-breaking phenolic antioxidants in vitro. J. Am.

Chem. Soc. 1981, 103, 6472-6477.

49. Yamauchi, R.; Ozaki, K.; Shimoyamada, M.; Kato, K., Iron-catalyzed reaction products of alpha-tocopherol with 1-palmitoyl-2-linoleoyl-3-sn-phosphatidylcholine

(13S)-hydroperoxide. Chem. Phys. Lipids 2002, 114, (2), 193-201.

156 50. Yamauchi, R.; Yagi, Y.; Kato, K., Oxidation of alpha-tocopherol during the

peroxidation of dilinoleoylphosphatidylcholine in liposomes. Biosci. Biotech. Biochem.

1996, 60, (4), 616-20.

51. Zhang, R.; Brennan, M. L.; Shen, Z.; MacPherson, J. C.; Schmitt, D.; Molenda, C. E.;

Hazen, S. L., Myeloperoxidase functions as a major enzymatic catalyst for initiation of

lipid peroxidation at sites of inflammation. J. Biol. Chem. 2002, 277, (48), 46116-22.

52. Poliakov, E.; Brennan, M. L.; Macpherson, J.; Zhang, R.; Sha, W.; Narine, L.;

Salomon, R. G.; Hazen, S. L., Isolevuglandins, a novel class of isoprostenoid derivatives,

function as integrated sensors of oxidant stress and are generated by myeloperoxidase in

vivo. Faseb J. 2003, 17, (15), 2209-20.

53. Christen, S.; Woodall, A.; Shigenaga, M.; Southwell-Keely, P.; Duncan, M.; Ames,

B., γ-tocopherol traps mutagenic electrophiles such as NOx and complements

α-tocopherol:Physiological implications. Proc. Natl. Acad. Sci. U. S. A. 1997, 94,

3217-3222.

54. Jiang, Q.; Wong, J.; Fyrst, H.; Saba, J. D.; Ames, B. N., gamma-Tocopherol or

combinations of vitamin E forms induce cell death in human prostate cancer cells by

interrupting sphingolipid synthesis. Proc. Natl. Acad. Sci. U. S. A. 2004, 101, (51),

17825-30.

55. Wechter, W. J.; Kantoci, D.; Murray, E. D., Jr.; D'Amico, D. C.; Jung, M. E.; Wang,

W. H., A new endogenous natriuretic factor: LLU-alpha. Proc. Natl. Acad. Sci. U. S. A.

1996, 93, (12), 6002-7.

157 56. Jiang, Q.; Elson-Schwab, I.; Courtemanche, C.; Ames, B. N., gamma-tocopherol and its major metabolite, in contrast to alpha-tocopherol, inhibit cyclooxygenase activity in macrophages and epithelial cells. Proc. Natl. Acad. Sci. U. S. A. 2000, 97, (21), 11494-9.

57. Jiang, Q.; Ames, B. N., Gamma-tocopherol, but not alpha-tocopherol, decreases proinflammatory eicosanoids and inflammation damage in rats. Faseb J. 2003, 17, (8),

816-22.

58. Devaraj, S.; Jialal, I., Failure of vitamin E in clinical trials: is gamma-tocopherol the answer? Nutr. Rev. 2005, 63, (8), 290-3.

59. Himmelfarb, J.; Hakim, R. M., Oxidative stress in uremia. Curr. Opin. Nephrol.

Hypertens. 2003, 12, (6), 593-8.

60. Cristol, J. P.; Canaud, B.; Rabesandratana, H.; Gaillard, I.; Serre, A.; Mion, C.,

Enhancement of reactive oxygen species production and cell surface markers expression due to haemodialysis. Nephrol. Dial. Transplant 1994, 9, (4), 389-94.

61. Satoh, M.; Yamasaki, Y.; Nagake, Y.; Kasahara, J.; Hashimoto, M.; Nakanishi, N.;

Makino, H., Oxidative stress is reduced by the long-term use of vitamin E-coated dialysis filters. Kidney Intern. 2001, 59, (5), 1943-50.

62. Miyazaki, H.; Matsuoka, H.; Itabe, H.; Usui, M.; Ueda, S.; Okuda, S.; Imaizumi, T.,

Hemodialysis impairs endothelial function via oxidative stress: Effects of vitamin E coated dialyzer. Circulation 2000, 101, 1002-1006.

63. Girndt, M.; Lengler, S.; Kaul, H.; Sester, U.; Sester, M.; Kohler, H., Prospective crossover trial of the influence of vitamin-E coated dialyzer membranes on T-cell activation and cytokine induction. Am. J. Kidney Dis. 2000, 35, 95-104.

158 64. Wanner, C.; Bahner, U.; Mattern, R.; Lang, D.; Passlick-Deetjen, J., Effect of

dialysis flux and membrane material on dyslipidaemia and inflammation in

haemodialysis patients. Nephrol. Dial. Transplant 2004, 19, (10), 2570-5.

65. Odetti, P.; Robaudo, C.; Valentini, S.; Gurreri, G.; Garibaldi, S.; Angeletti, S.;

Deferrari, G., Effect of a new vitamin E-coated membrane on glycoxidation during hemodialysis. Contrib. Nephrol. 1999, 127, 192-9.

66. Baragetti, I.; Furiani, S.; Vettoretti, S.; Raselli, S.; Maggi, F. M.; Galli, F.; Catapano,

A. L.; Buccianti, G., Role of Vitamin E-Coated Membrane in Reducing Advanced

Glycation End Products in Hemodialysis Patients: A Pilot Study. Blood Purif. 2006, 24,

(4), 369-376.

67. Odetti, P.; Traverso, N.; Monacelli, F.; Menini, S.; Vazzana, J.; Tasso, B.; Pronzato,

M. A.; Robaudo, C.; Deferrari, G., Vitamin E-coated filter decreases levels of free

4-hydroxyl-2-nonenal during haemodialysis sessions. Free Radic. Res. 2006, 40, (2),

207-12.

68. Sommerburg, O.; Zang, L.-Y.; van Kuijk, F., Simultaneous detection of carotenoids and vitamin E in human plasma. J. Chromatogr. B 1997, 695, 209-215.

69. Odetti, P.; Fogarty, J.; Sell, D. R.; Monnier, V. M., Chromatographic quantitation of plasma and erythrocyte pentosidine in diabetic and uremic subjects. Diabetes 1992, 41,

153-159.

70. Friedlander, M.; Wu, Y.; Elgawish, A.; Monnier, V., Early and Advanced

Glycosylation End Products: Kinetics of formation and clearance in peritoneal dialysis. J.

Clin. Invest. 1996, 97, 728-735.

159 71. Sell, D. R., and Monnier, V. M., Structural elucidation of a fluorescent cross link from human senescent extracellular matrix: implication of pentoses in the aging process. .

J. Biol. Chem. 1989, 264, 21597-21602.

72. Friedlander, M.; Witko-Sarsat, V.; Nguyen, A.; Wu, Y.; Labrunie, M.; Verger, C.;

Jungers, P.; Descamps-Latscha, B., The advanced glycation endproduct pentosidine and monocyte activation in uremia. Clin. Nephrol. 1996, 45, 379-382.

73. Friedlander, M.; Wu, Y.; Schulak, J.; Monnier, V.; Hricik, D., Influence of dialysis modality on plasma and tissue concentrations of pentosidine in patients with end-stage renal disease. Am. J. Kidney Dis. 1995, 25, (3), 445-451.

74. Salomon, R. G.; Sha, W.; Brame, C.; Kaur, K.; Subbanagounder, G.; O'Neil, J.; Hoff,

H. F.; Roberts, L. J., 2nd, Protein adducts of iso[4]levuglandin E2, a product of the isoprostane pathway, in oxidized low density lipoprotein. J. Biol. Chem. 1999, 274, (29),

20271-80.

160

Chapter 5

Identification of Carboxyethylpyrrole Phosphatidylethanolamine

Adducts in vitro and in vivo

161 5.1. Background

Retinal membrane contains the most highly unsaturated fatty acids found in vertebrate tissues. Over 50% of the total retinal fatty acids are unsaturated.1 Of all subcellular components of retina, the rod outer segment (ROS) is the most enriched in polyunsaturated fatty acid (PUFA) phospholipid esters, among which docosahexaenoic acid (DHA) phospholipids predominate (see Table 5.1). The major phospholipids in ROS are phosphatidylethanolamines (PEs) and phosphatidylcholines (PCs), which comprise

37.6% and 32.5% of total lipids respectively.

Table 5.1. Distribution of the fatty acids of PE and PC in human ROS (as mol% of total fatty acid).

Phosphatidylethanolamine Phosphatidylcholine (PC)

(PE)

Palmitic acid (16:0) 13.3 32.5

Linoleic acid (18:2, ω6) 0.3 1.1

Arachidonic acid (20:4, 3.5 4.7

ω6)

Docosahenaenoic acid 34.2 19.5

(22:6, ω3)

% saturation 52.3 48.9

% unsaturation 43.1 27.3

162 In retina, light and oxygen are essential for vision. However, the formation of reactive

oxygen species, which leads to photochemical damage, is also favored by light and

oxygen.2 Retina photoreceptor rod outer segments (ROS) consist of a stack of hundreds of densely packed membrane disks that are renewed continually. New disks are generated

in the rod inner segment and migrate to the base of the rod outer segment, and old disks

are detached from the end of the rod outer segment where they are deformed, engulfed,

and displaced within the retinal pigment epithelium (RPE).3, 4 This process is believed to be related to oxidative modifications induced by photogenerated radicals.5 The high

levels of oxygen consumption and irradiation received by retina is conceivably

responsible for the generation of reactive oxygen species through photosensitizers.2, 6 A

large number of chromophores may serve as potential sensitizers such as the visual

pigments (11-cis-retinal-protein complexes), melanin and lipofuscin, flavins and

flavoproteins, and the macular pigment.2 Lipofuscin (LF) is an ill-defined conglomerate

of lipids, metals, organic molecules, and biomolecules that commonly fluoresce at 360 to

470 nm. Lipofuscin-like substances were observed by a reaction of lipids containing

primary amines, with either an aldehyde or a ketone, in the presence of a peroxide.7, 8

Lipofuscin serves to facilitate generation of reactive oxygen species that contribute to

age-related decline of retinal pigment epithelium function and blue light damage.9 It has

been confirmed that age-related macular degeneration (AMD) is significantly correlated

(P = 0.01) with lipofuscin pigment accumulation in the basal lamina and Bruch’s

membrane cells of the retina.10

163 The structure of fluorophores in lipofuscin granules remained unknown until the documentation of A2E.11 The name A2E stemmed from the characterization that this unusual bis-retinoid is derived from 2 molecules of retinal and one molecule of ethanolamine.12 A2E is biosynthesized in ROS from all-trans-retinal and PEs in the form of a phospholipid bisretinoid adduct (A2-PE), which is subject to hydrolytic cleavage by phospholipase D to deliver A2E.13 A2E accumulates in the RPE as the consequence of the accumulation of large amounts of all-trans-retinal which failed to be efficiently reduced to all-trans-retinol.14 Upon irradiation (430 nm), A2E acts as a photosensitizer to release singlet oxygen and other reactive oxygen species, which could trigger the epoxidation and peroxidation of polyunsaturated fatty acids and therefore contribute to photooxidative damage such as modifications of DNA and protein.15-18 It is also conceivable that A2E could induce cell apoptosis after exposure to blue light (480 nm), and exert a detergent-like perturbation of cell membranes.19-21 A2E was previously suggested to exercise toxic effects on RPE cells by directly damaging lysosomal function and structure.22 Extralysosomal RPE cell structures were also proposed to be severely harmed by A2E during the pathogenesis of AMD. The identification of A2E was a breakthrough that exemplified the pathological potential of lipid-modified PEs.

The primary amino groups of PEs, as well as proteins, are potential targets of covalent modifications by carbohydrates and lipid peroxidation products. In 1993, Bucala et al.23 first proposed that glucose would modify the amino group of PEs. Subsequent in vitro studies24, 25 confirmed the formation of glucose-modified PE through an unstable Schiff

164 base that rearranges to a PE-linked α-aminoketone (Amadori) product. The existence of

glycated aminophospholipids in human red blood cells and plasma was further

documented by Ravandi et al.26 Another glycation adduct, carboxymethyl-PE (CME) was identified in red blood cell membranes and fasting urine, although the amount of CME did not show statistically significant differences for control versus diabetic subjects.27

PEs modified by lipid peroxidation products were also reported in recent years.

Malonaldehyde, an aldehydic degradation product from lipid peroxidation, was proposed to react with amino groups in phosphatidylethanolamine in a model study to generate fluorescent Schiff’s bases with a 1-amino-3-imino-propene structure.28 It was further

documented by Esterbauer, et al., that the reaction between 4-hydroxynonenal and

phosphatidylethanolamine led to the formation of a fluorescent chromophore identical

with the fluorophore formed in peroxidizing microsomes and mitochondria. Tsuji et al.29

characterized the formation of N-(hexanoyl)phosphatidylethanolamine (HEPE), in the

reaction of egg PE with 13-hydroperoxyoctadecadienoic acid. A Michael adduct,

generated by covalent binding of 4-HNE to PEs, was elucidated to be a poor substrate of

30 secreted phospholipase A2 and phospholipase D. In an in vitro reaction, phosphatidyl

ethanolamine-derived pyrroles 5.3 and 5.4 were identified as products from incubation of phosphatidylethanolamine with 4,5-(E)-epoxy-2-(E)-heptenal (5.1) followed by GC/MS or HPLC/MS (Scheme 5.1).31 By comparing the ability of 4-HNE, 4-hydroxydodeca-

(2E,6Z)-dienal (4-HDDE), and 4-hydroxy-2E- hexenal (4-HHE) from n-3 fatty acids to

covalently modify various ethanolamine phospholipids (PEs), chosen for their biological

165 relevance, it was concluded that aldehydes that are more hydrophobic generate more

adducts with the PEs.32 Moreover, the aldehydes exhibited stronger reactivity toward

32 PEs having unsaturated fatty acids compared with saturated ones. Levuglandin E2 stereoisomers, generated by the free radical induced peroxidation of arachidonic acid via the isoprostane pathway, were also shown to covalently modify PEs in vitro to deliver pyrrole and Schiff base adducts.33

O O R-NH2 O Et Et + Et H NR N N R R OH 5.1 5.2 5.3 5.4

Scheme 5.1. 4,5-(E)-Epoxy-2-(E)-heptenal reacts with phosphatidylethanolamine

(R-NH2) to produce pyrrole derivatives.

Modified PEs were shown to have some biological activities. Oxidative modifications

of the LDL-associated PEs stimulate a pronounced prothrombotic response by increasing

the activity of the platelet prothrombinase complex.34 The concentration of

p-hydroxyphenylacetaldehyde (pHA)-modified phospholipid, that is generated from this

product of L-tyrosine oxidation and the amino group of PEs, is strikingly elevated in

LDL isolated from human atherosclerotic lesions.35 Synthetically prepared

Amadori-glycated PE triggered lipid peroxidation when this Amadori-PE was incubated

with linoleic acid in the presence of Fe3+ in a micellar system.36

Previously, research in our group has shown that oxidative cleavage of retinal

phospholipids containing DHA produces reactive electrophilic 4-hydroxy-7-oxohept

166 -5-enoates that convert the primary amino group of protein lysyl residues into

2-(ω-carboxyethyl)pyrrole (CEP) derivatives.37 Evidence is accumulating showing that

CEPs are closely associated with age-related macular degeneration (AMD).38, 39 In view

of the reactivity of the primary amino group of PEs and the abundance of DHA in brain

and retina, we anticipate that DHA-derived oxidatively truncated phospholipids containing reactive electrophilic 4-hydroxy-7-oxohept-5-enoates convert the primary amino group of PEs into carboxyethylpyrrole phosphatidylethanolamine adducts

(CEP-PEs). In this chapter, we identified and quantified the CEP-PEs in lipid extracts from light promoted oxidation of bovine retina. Pilot studies on plasma from an age-related macular degeneration (AMD) patient and a normal control show that carboxyethylpyrrole phosphatidylethanolamine adducts are endogenously generated in vivo. The formation of these adducts in vivo could be involved in the pathogenesis of

AMD and other diseases associated with oxidative injury.

167 5.2. Results and Discussion

5.2.1. Syntheses of authentic samples. Authentic samples of CEP-PE and lysoCEP-PE

were synthesized by reaction of DOHA-Fm with 1-palmityl-2-oleyl-sn-glycero-3-

phosphoethanolamine (POPE) or 1-palmitoyl-2-hydroxy-sn-glycero-3-phospho-

-ethanolamine (lysoPE) followed by deprotection of the intermediate Fm esters with

DBU (Scheme 5.2). The synthesis of authentic standards and their characterization by

high resolution mass spectrometry (HRMS) and multinuclear NMR were described in

Chapter 2.

O CO POPE 76% R O P O O DOHAFm + or O lysoPE OH N TEA, CHCl3 O C14H29 CEPFm-PE, R = oleoyl O lysoCEPFm-PE, R = H O CO R O P O OH 90% O OH N DBU, CHCl 3 O C14H29 CEP-PE, R = oleoyl O lysoCEP-PE,R= H Scheme 5.2. Synthesis of CEP-PE and lysoCEP-PE.

5.2.2. Immunoreactivity of authentic CEP-PE. Like CEP-dipeptide, CEP-PE has a

CEP epitope, that is expected to be recognized by anti-CEP antibody. A competitive

ELISA was performed to characterize the competitive binding of CEP-PE to anti-CEP-KLH polyclonal antibody. CEP-CEO (chicken egg ovalbumin) was used as a coating agent and a standard, CEP-PE was used as an inhibitor. As shown in Figure 5.1,

CEP-PE is cross-reactive with anti-CEP antibody, although the binding of CEP-PE to

168 anti-CEP antibody is not as strong as that of CEP-CEO, perhaps due to the low molecular

weight of the former.

100

80

60

40 Absorbance (%max) 20

0

10-1 100 101 102 103 104 Inhibitor (pmol/mL)

Figure 5.1. Inhibition curves showing crossreactivity of the anti-CEP-KLH antibody for

CEP-CEO (y) and CEP-PE (ƒ) against CEP-CEO as coating agent.

5.2.3. Identification of CEP-PE in oxidized lipid extracts. It has been reported that light damage promotes the peroxidation of DHA in retina.40 We postulated that irradiation of a lipid extract from retina would produce CEP modified PEs through oxidation of DHA to HOHA, that would, in turn, covalently bind with the primary amino group of PEs. Oxidation of lipid vesicles containing 50% retinal lipid extract and 50%

POPC was promoted with UV light of 350 nm. Butylated hydroxytoluene (BHT) was added after 33 h to prevent further autoxidation of the lipid samples. Lipids were extracted using the Bligh and Dyer method,41 dried under a nitrogen stream at room

169 temperature, sealed under argon, and stored in the dark at -80°C until they were analyzed.

The extracts were analyzed by LC/MS, and preliminary identification of products was achieved by comparing retention times with those of authentic samples. Negative ion

ESI-MS/MS analysis of CEP-PE yielded both parent and daughter ions which were used as the specific mass transition ion pairs in LC-MS/MS analysis. The multiple reaction monitoring (MRM) transitions used to identify the CEP-PE were the mass-to-charge ratios (m/z) for the molecular ion [M-H]- and its daughter ions, which correspond to the fragments listed in Figure 5.2. A daughter scan spectrum of CEP-PE is presented in

Figure 5.3. MRM chromatograms of CEP-PE are presented in Figure 5.4. Oxidized lipid extracts from bovine retina produced the expected CEP-PE. A single peak with a retention time that is identical to that of standard was present in the MRM chromatogram of oxidized lipid in each parent/daughter ion pair channel (Figure 5.5).

5.2.4. Identification of CEP-PE in lipid extracts that had not been oxidized in vitro.

Lipid extracts from bovine retina were dissolved in methanol and analyzed by LC-MS

using the negative ion MRM mode. The specific parent-daughter ion pairs were

monitored and a single peak with a retention time that is identical to that of the standard

is present in the MRM chromatogram monitoring the 838.3 → 255.4 and 838.3 → 391.2

transitions (Figure 5.6).

170 COO Parent ion O O O : 838.5 P N O O OH C8H17 O C14H29 O

O O O P O OH O Daughter ion: 673.6 C8H17 O C14H29 O O O O P Daughter ion: 417.2 O O OH

C8H17 O O P O OH Daughter ion: 391.2 C15H31 O O O

O Daughter ion: 281.2

C8H17

O Daughter ion: 255.2 O O O HO P Daughter ion: 152.9 O OH

Figure 5.2. Proposed MRM fragments of CEP-PE and corresponding m/z.

Relative Intensity (%) m/z Figure 5.3. Daughter scan of CEP-PE.

171 Relative Intensity (%) Relative Intensity

Figure 5.4. MRM chromatograms of CEP-PE.

172 Relative Intensity (%) Relative Intensity

Figure 5.5. MRM of lipid extracts from bovine retina after UV light-promoted oxidation.

A single peak with a retention time that is identical to that of authentic standard CEP-PE was present in the MRM chromatogram of oxidative lipid at each parent/daughter ion pair channel.

173

Relative Intensity (%) Relative Intensity

Figure 5.6. MRM of bovine retinal extracts that had not been oxidized in vitro. A single peak with a retention time that is identical to that of authentic CEP-PE is present in the

MRM chromatogram monitoring the 838.3 → 255.4 and 838.3 → 391.2 transitions.

174 5.2.5. Quantification of CEP-PE in bovine retina. Calibration curves for quantitative

analyses of CEP-PE were constructed by adding a fixed amount of internal standard

1-myristyl-2-hydroxy-sn-glycero-3-phosphatidylethanolamine (M-PE) into various amounts of authentic CEP-PE samples. LC-MS analysis was performed and peak area ratio vs analyte weight ratio was plotted. Calibration curves of CEP-PE and equations describing the calibration curves are presented in Figure 5.7. Samples containing lipid extracts and a fixed amount of internal standard (M-PE), that were or were not exposed to light-promoted oxidation, were analyzed by LC-MS. There was 60 pg CEP-PE in 1.0 μg

of lipid extract with light-promoted oxidation and 0.7 pg CEP-PE in 1.0 μg of lipid extract without oxidation.

8 y = 1.2168x - 0.1121

E 7 R2 = 0.9986 6 5 4 3 2 1 Peak AreaRatio (Analyte/MP 0 01234567 Analyte (w)/MPE (w)

Figure 5.7. Standard curve and calibration equation for CEP-PE.

5.2.6. Identification of lysoCEP-PE in light-promoted oxidized lipid extracts. In glycerophospholipids, the sn-1 position is usually occupied with saturated C16 or C18

175 fatty acids, while the sn-2 position may have a variety of saturated or unsaturated fatty acyls. Phospholipase A2 (PLA2) enzymes catalyze hydrolysis of sn-2 acyl chains from membrane phospholipids in human tissue and cells to liberate free fatty acids and lysophospholipids.42 To simplify the analysis of oxidative phospholipid modifications, we explored monitoring of lysoCEP-PE in retina samples. A complex mixture of CEP modified PEs can be converted into a simpler mixture by the action of PLA2. An authentic standard sample of lysoCEP-PE was obtained by reaction of DOHA-Fm with lysoPE followed by deprotection of the intermediate Fm ester with DBU. We also converted authentic CEP-PE to lysoCEP-PE by treating it with PLA2. This sample of lysoCEP-PE was characterized by HRMS and NMR. Negative ion ESI-MS/MS analysis of lysoCEP-PE showed parent and daughter ions that were used as the specific mass transition ion pairs for LC-MS/MS analysis. The MRM transitions used to detect the lysoCEP-PE were the mass-to-charge ratios (m/z) for the molecular ion [M-H]- and its daughter ions, which correspond to the fragments listed in Figure 5.8. A daughter scan spectrum of lysoCEP-PE is presented in Figure 5.9. It should be noted that the fragment that represents m/z = 317.98 has the specific carboxyethylpyrrole group in it. The detection of this fragment thus provides presumptive evidence that irradiated lipid extracts from bovine retina contain lysoCEP-PE (vide infra). Chromatograms of authentic standard lysoCEP-PE are presented in Figure 5.10. A lipid extract from bovine retina was converted into vesicles and irradiated with UV light (350 nm) for 33 h, and then extracted and analyzed by LC/MS as described above. A single peak with a retention time that is

176 identical to that of authentic standard is present in the MRM chromatogram of oxidized lipid in each parent/daughter ion pair channel (Figure 5.11).

COO

O O P N Parent ion: 574.07 HO O OH

O C14H29 O O P O HO O OH Daughter ion: 409.29

O C14H29 O COO

O P O Daughter ion: 317.98 N HO O OH

O Daughter ion: 255.2 O O O P Daughter ion: 152.9 O OH HO Figure 5.8. Proposed fragments of lysoCEP-PE and corresponding m/z. Relative Intensity (%)

m/z Figure 5.9. Daughter scan of lysoCEP-PE.

177 5.15 100 574.1 > 409.3

0 5.15 100 574.1 > 317.98

0

100 5.16 574.1 > 255.2 Relative Intensity (%) Relative Intensity

0

100 5.16 574.1 > 152.89

0 0 Time (minute) 14 Figure 5.10. MRM chromatograms of lysoCEP-PE. Pure lysoCEP-PE (10 ng/μL in methanol) was injected onto a Shimadzu LC-10AD HPLC system, eluting with 1 mM ammonium acetate in 20% acetonitrile and using a linear gradient to 100% acetonitrile over 3 minutes, followed by 100% acetonitrile for 10 minutes.

178 574.07 > 409.29 100 5.16

0 5.16 100 574.07 > 317.98

0 5.16 574.07 > 255.22

Relative Intensity (%) Relative Intensity 100

0 5.16 100 574.07 > 152.89

0 14 0 Time (minute) Figure 5.11. MRM of bovine retinal lipids after UV-promoted oxidation. Lipid extracts

(50 ng/μL in methanol) was injected onto a Shimadzu LC-10AD HPLC system as described in Figure 5.10. A single peak with a retention time that is identical to that of authentic lysoCEP-PE was present in the MRM chromatogram of oxidized lipid in each parent/daughter ion pair channel.

179 5.2.7. Identification of lysoCEP-PE in bovine retina. Bovine retinal lipid that had not been modified by UV-promoted oxidation was also analyzed through LC-MS/MS. Its

MRM chromatogram did not display these characteristic peaks. We also treated these

lipid extracts with PLA2 to convert a mixture of CEP-PEs into a simpler mixture of

lysoCEP-PEs and monitored lysoCEP-PE using the MRM mode by LC-MS/MS.

However, the results did not show any characteristic peaks for lysoCEP-PE in the MRM

chromatogram. This may be because lipids from a different retina than that described

above (P.170) were used and they had no CEP-PE. Alternatively, the presence of EDTA

may have interfered with the action of PLA2. This issue should be resolved by further

studies.

5.2.8. Identification of lysoCEP-PE in human plasma. Lipid extracts from plasma

samples of an AMD patient and a normal control were analyzed by LC-MS/MS. A single

peak with a retention time that is identical to authentic lysoCEP-PE is present in the

MRM chromatogram in each parent/daughter ion pair channel (Figures 5.12-5.14).

180 100 8.80 574.07 > 409.29

0

100 8.80 574.07 > 317.98

0 8.84

Relative Intensity (%) Relative Intensity 100 574.07 > 255.22

0 8.84 100 574.07 > 152.89

0 0 15 Time (minute)

Figure 5.12. MRM chromatograms of authentic lysoCEP-PE. Pure lysoCEP-PE (50 ng) was injected onto a Waters 2790 HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes. Note: This is a different system than Figures 5.10-5.11 and retention times are, therefore, different.

181 100 8.84 574.07 > 409.29

0

100 8.88 574.07 > 317.98

0

Relative Intensity (%) Relative Intensity 8.88 100 574.07 > 255.22

0

100 8.88 574.07 > 152.89

0

0 Time (minute) 18

Figure 5.13. MRM chromatograms of lipid extracts from a plasma sample of a normal control. Lipid extracts (150 μg in 20 μL methanol) was injected onto a Waters 2790

HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes.

182 100 8.77 574.07 > 409.29

0 100 8.81 574.07 > 317.98

0

Relative Intensity (%) Relative Intensity 100 8.77 574.07 > 255.22

0 8.77 100 574.07 > 152.89

0 0 Time (minute) 18 Figure 5.14. MRM chromatograms of lipid extracts from a plasma sample of an AMD patient. Lipid extracts (100 μg in 20 μL methanol) was injected onto a Waters 2790

HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes.

183 5.2.9. Quantification of lysoCEP-PE in bovine retina and human plasma. Calibration

curves for quantitative analyses of lysoCEP-PE were constructed by adding a fixed

amount of internal standard 1-myristyl-2-hydroxy-sn-glycero-3-phosphatidyl

ethanolamine (M-PE) into various amounts of authentic lysoCEP-PE samples. LC-MS

analysis was performed and peak area ratio vs analyte weight ratio was plotted.

Calibration curves of lysoCEP-PE and equations describing the calibration curves are

presented in Figure 5.15. Lipid extracts samples containing a fixed amount of internal

standard (M-PE) were analyzed by LC-MS. There was 260 pg lysoCEP-PE in 1.0 μg of

lipid extract with light-promoted oxidation. No lysoCEP-PE was detected in lipid extract

that had not been subjected to light-promoted oxidation. There was 1.2 ng lysoCEP-PE in

1 mL of plasma from a normal control and 1.3 ng lysoCEP-PE in 1 mL of plasma from

an AMD patient. These levels are virtually identical.

7 E 6 y = 2.7858x R2 = 0.9995 5 4 3 2 1 Peak Area Ratio (Analyte/MP 0 00.511.522.5 Analyte (w)/MPE (w)

Figure 5.15. Standard curve and calibration equation of authentic lysoCEP-PE.

184 5.3. Conclusions

Recent studies have strongly implicated reactive aldehydes derived from lipid peroxidation could be key mediators of oxidant injury in view of their potency to covalently modify proteins, lipids, and DNA.34, 43 Modification of aminophospholipids in

plasma membrane might disturb the phospholipid membrane distribution, alter membrane

fluidity, and damage membrane organization and function.44 HNE and isolevuglandins

have been documented to react with PEs. In view of the striking abundance of DHA and

PEs in retinal photoreceptor outer segment membranes, we explored the possibility that

CEP-PE adducts are generated in retina, presumably through oxidation of

DHA-containing phospholipids to HOHA-containing phospholipids, followed by

covalent binding to the primary amine of PEs and partial phospholipolysis. Using an

authentic standard CEP-PE, 60 pg CEP-PE in 1.0 μg lipid extract and 0.7 pg CEP-PE in

1.0 μg of lipid extract were detected and quantified in bovine retinal extracts that had and

had not been modified by UV light-promoted oxidation respectively. In addition, 260 pg of a CEP-modified lysophospholipid, lysoCEP-PE was identified and quantified in 1.0 μg

of bovine retinal extracts that had been modified by UV light-promoted oxidation. In a pilot study, similar levels, about 1.2 ng/mL, of lysoCEP-PE were detected in plasma

samples from a normal control and an AMD patient. While further studies on a large

number of individuals may reveal a significant difference between AMD and normal

individuals, it seems unlikely that a large difference will be found.

To simplify the analysis of oxidative phospholipid modifications, I treated the lipid

185 extract with PLA2. When I extracted lipid from bovine retina, I added Na2EDTA and

2+ BHT to the lipid extract to prevent oxidation. In view of the fact that PLA2 requires Ca

as a cofactor, I added extra Ca2+ solution to the system. Nevertheless, I did not detect

lysoCEP-PE in lipid extracts from non-oxidized bovine retina before or after treatment

with PLA2. There are two possible issues: 1, In the presence of Na2EDTA, the activity of

PLA2 was blocked and therefore no CEP-PE was converted to lysoCEP-PE. 2. There was

no HODA-PCs present in the retina sample used for this experiment. The following

experiments may clarify the issues. Add authentic CEP-PE to lipid extracts, then treat the

2+ mixture with PLA2 plus extra Ca solution to see if CEP-PE converts to lysoCEP-PE

under these conditions. Another possible solution may be to replace Na2EDTA with

CaEDTA that is expected to chelate Fe2+ or Cu1+ by exchange for Ca2+ to prevent oxidation of lipid extracts. Then we can treat real samples containing CaEDTA with PLA2 and analyze through LC-MS to check for the generation of lysoCEP-PE. As a control, we will treat CEP-PE with PLA2 under these same conditions.

186 5.4. Experimental Procedures

Materials and Methods

1-Palmityl-2-oleyl-sn-glycero-3-phosphatidylethanolamine (PO-PE),

1-palmityl-2-hydroxy-sn-glycero-3-phosphatidylethanolamine (Lyso-PE) and

1-myristyl-2-hydroxy-sn-glycero-3-phosphatidylethanolamine (M-PE) were obtained

from Avanti Polar Lipids (Alabaster, AL).

Phospholipase A2 from porcine pancreas was obtained from Sigma (Cat. P0861).

ELISA Characterization of competitive binding of CEP-PE to anti-CEP antibody.

To test the immunoreactivity of authentic CEP-PE to anti-CEP-KLH polyclonal antibody,

a competitive ELISA was performed. CEP-PE vesicles were prepared by mixing CEP-PE

(50 μg) and PO-PC (450 μg) in PBS buffer (80 μL, 10 mM, pH 7.4). The mixture was

sonicated for 5 min and made into unilamellar vesicles by extrusion (4 passes) using an

Avanti Mini-Extruder (Avanti Polar Lipids, Inc.).45 CEP-CEO was used as a coating

agent and a standard. The CEP-CEO was synthesized as described in Chapter 2. The

standard was prepared by diluting a 251.4 μmol/L CEP-CEO solution to 2514 nmol/L

with PBS (10 mM, pH 7.4). A serial dilution factor of 0.2 was used, and up to eight serial dilutions of CEP-PE vesicles and CEP-CEO standard were run. CEP-CEO (100 µL,

125.7 nmol/L) solution was coated onto each well of the plate. The ELISA was carried out as described in Chapter 2. The results are summarized in Table 5.2 and Table 5.3 and shown graphically in Figure 5.1.

187 Table 5.2. ELISA data for CEP-PE in Figure 5.1.

Pmol/mL Absorbance % Absorbance Parameters Curve Fit

5200. 0.1840 23.2911 a = 100.00 25.5699

1040. 0.2280 28.8608 b = 0.8649 27.0222

208. 0.2400 30.3797 c = 1169.1756 31.2217

41.6 0.3770 47.7215 d = 25.5178 44.1505

8.32 0.5020 63.5443 68.1990

1.664 0.7190 91.0127 88.3608

0.3328 0.7880 99.7468 96.7218

0.0666 0.7940 100.5063 99.1573

Table 5.3. ELISA data for CEP-CEO in Figure 5.1.

Pmol/mL Absorbance % Absorbance Parameters Curve Fit

2514. 4.0000e-3 0.3035 a = 100.00 0.3105

502.8 0.0200 1.5175 b = 1.1669 0.6099

100.56 0.0360 2.7314 c = 4.0079 2.5249

20.112 0.1520 11.5326 d = 0.2563 13.4347

4.0224 0.6780 51.4416 50.0232

0.8045 1.1330 85.9636 86.7251

0.1609 1.2540 95.1442 97.7126

0.0322 1.3180 100.0000 99.6434

188 Extraction of Phospholipids from Retina. Bovine retinas were harvested from 2 normal cows. To preclude contamination by blood and to prevent in vitro oxidation, the retinas were rinsed with saline antioxidant cocktail [saline PBS, pH 7.4, containing 2 mM

ethylenediaminotetraacetic acid (Na2EDTA) and 100 μM butylated hydroxytoluene

(BHT)]. Lipid extraction was performed using the following procedure. Methanol (1 mL)

and 0.5 M HCl (5 μL) were added to the retina. The tissue was immediately homogenized

manually using a stainless steel pestle coated with Teflon. Then chloroform (1 mL) was

added. The mixture was vortexed, centrifuged at 3000 rm, 4 °C for 3 minutes. The lower

organic layer was carefully collected into a brown vial. The aqueous layer was extracted

with chloroform (2 x 1 mL) and the organic layer was combined and dried under a stream

of argon and stored under argon in an amber vial at -80 °C before being analyzed by

LC-MS/MS.

Light-promoted lipid oxidation in vitro. Unilamellar vesicles were prepared as follows.

Lipid extract (1 mg) and PO-PC (1 mg) were suspended in PBS buffer (1 mL, 10 mM,

pH 7.4). The mixture was vortexed for 1 minute, sonicated for 5 minutes and passed

through an extruder. The vesicles were kept in a quartz tube placed in the center of a

Rayonet photochemical reactor with three 80 W low-pressure mercury UV (350 nm)

Rayonet lamps (Southern New England Ultraviolet, Midtown, CT) at room temperature

for 33 h. Lipid extraction was then performed according to Bligh and Dyer41, and the

samples were dried, stored, and sealed, in vials, under argon, at -80 ºC.

189 Bligh and Dyer method for lipid extraction. Chloroform and methanol (3.75 mL,

CHCl3/MeOH = 1:2, v/v) was added to lipid vesicles in PBS buffer (1 mL). The mixture was vortexed vigorously. Chloroform (1.25 mL) was added and the mixture was vortexed vigorously again. Finally H2O (1.25 mL) was added and the system was vortexed well.

The resulting mixture was centrifuged at 1000 RPM for 5 minutes at room temperature to give two phases. The bottom organic phase was withdrawn very carefully through a

Pasteur pipette to avoid disturbing the interface or upper phase. The lipid was concentrated by rotary evaporation and the residue dried fully under a stream of argon.

Synthesis of lysoCEP-PE by treating authentic CEP-PE with PLA2. CEP-PE (14 mg,

0.017 mmol) was dissolved in THF (50 μL). CaCl2 (5 mM, 70 μL) in pH 7.4 PBS solution (10 mM, 1 mL) was added. The system was sonicated for 2 min followed by the

addition of PLA2 (10,000 unit/mL, 10 μL) and then incubated in 37 °C for 5 h. TLC

(CHCl3 : MeOH : H2O = 65 : 25 : 4, Rf = 0.3) showed the reaction was completed. The mixture was extracted with CHCl3/MeOH (2 : 1, v/v, 1 mL x 3) and the organic layer was concentrated to afford crude product. Flash chromatography (CHCl3 : MeOH : H2O = 65 :

1 25 : 4) provided pure lysoCEP-PE (6 mg, 65%). H NMR (CD3OD : CDCl3 = 1 : 1, 400

MHz), δ 6.60 (m, 1H), 5.96 (m, 1H), 5.83 (m, 1H), 4.10-4.0 (5H), 3.74-3.6 (m, 2H),

3.6-3.5 (m, 2H), 2.8-2.9 (m, 2H), 2.68-2.65 (m, 2H), 2.32 (t, J = 6.8 Hz, 2H), 1.57 (m,

2H), 1.23 (24H), 0.85 (t, J = 7.2 Hz, 3H).

LC/MS/MS analysis and quantification of CEP-PE in lipids from bovine retina.

A. Analysis of standard. LC-MS analysis was performed on a Quattro Ultima mass

190 spectrometer (Micromass, Wythenshawe, United Kingdom) equipped with an

electrospray ionization (ESI) probe interfaced with a Shimadzu LC-10AD (Kyoto, Japan)

HPLC system. A standard curve for CEP-PE was generated by incorporating a fixed

amount of internal standard (M-PE) and varying levels of pure CEP-PE, and plotting

peak area ratio vs analyte weight ratio. M-PE (10 μL, 1 ng/μL) was added to 90 μL each of various concentrations (15 pg/μL, 30 pg/μL, 60 pg/μL, 120 pg/μL, 240 pg/μL, 480 pg/μL) of CEP-PE in methanol. Each analyte (30 μL) mixed with internal standard was chromatographed on a BetaBasic C18 column (20 x 2.1 mm, 5 μ, Thermo Electron

Corporation, Part NO. 71505-022150) at a flow rate of 0.2 mL/min, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 10 min, followed by 100% methanol for 10 min. Mass spectrometric analysis was performed online using an ESI tandem mass spectrometer in the negative ion multiple reaction monitoring (MRM) mode (cone energy, 60 V; collision energy, 40 eV). The total ion current was measured in the mass range of m/z 200-1000 in the negative ion mode. A 3.1 kilovolt potential was applied to the electrospray capillary. The optimal collision energy to obtain the most intense daughters was determined to be 40 eV. The MRM transitions used to detect the CEP-PE were the mass-to-charge ratios (m/z) for the molecular ion

[M-H]- and its daughter ions, which correspond to the fragments listed in Figure 5.2. The

following mass transitions, m/z 838.3 → 391.2, 838.3 → 281.4, 838.3 → 255.4, 838.3 →

153 were monitored simultaneously. Chromatograms are displayed in Figure 5.4.

B. Analysis of bovine lipids. Bovine retinal lipids that had been oxidized (promoted by

191 UV irradiation) in vitro (500 μg) were dissolved in chloroform : methanol (1 : 1, 1 mL).

Bovine retinal lipids that had not been oxidized in vitro (500 μg)was dissolved in chloroform : methanol (1 : 1, 100 μL). To these solutions (90 μL) was added M-PE (10

μL, 1 ng/μL in methanol). The resulting mixture (30 μL) was analyzed by LC-MS/MS using the same HPLC gradient and MS/MS conditions as described above for pure

CEP-PE. The mass transitions, m/z 838.3 → 391.2, 838.3 → 281.4, 838.3 → 255.4,

838.3 → 153 were monitored simultaneously. Chromatograms of lipid extracts with and without oxidation are displayed in Figures 5.5 and 5.6, respectively. The amount of

CEP-PE was 60 pg in 1 μg lipid extracts from oxidized bovine retina and 0.7 pg in 1 μg lipid extracts from bovine retina without light-promoted oxidation.

LC/MS/MS analysis and quantification of lysoCEP-PE generated in light-promoted oxidation of bovine retina. A standard curve for lysoCEP-PE was generated by incorporating a fixed amount of internal standard (M-PE) and varying levels of pure lysoCEP-PE, and plotting peak area ratio vs analyte weight ratio. M-PE (10 μL, 1 ng/μL in methanol) was added to 90 μL each of various concentrations (5 pg/μL, 10 pg/μL, 20 pg/μL, 40 pg/μL, 80 pg/μL, 250 pg/μL) of lysoCEP-PE in methanol. Each analyte (30 μL) mixed with internal standard was chromatographed on a BetaBasic C18 column (20 x 2.1 mm, 5 μ, Thermo Electron Corporation, Part NO. 71505-022150) at a flow rate of 0.2 mL/min, eluting with 1 mM ammonium acetate in 20% acetonitrile and using a linear gradient to 100% acetonitrile in 3 min, followed by 100% acetonitrile for 10 min. Mass spectrometric analysis was performed online using an ESI tandem mass spectrometer in

192 the negative ion multiple reaction monitoring (MRM) mode (cone energy, 45 V; collision

energy, 24 eV). The total ion current was measured in the mass range of m/z 200-1000 in

the negative ion mode. A 3.77 kilovolt potential was applied to the electrospray capillary.

The optimal collision energy to obtain the most intense daughters was determined to be

24 eV. The following mass transitions, m/z 574.07 → 409.29, 574.07 → 317.98, 574.07

→ 255.22, 574.07 → 152.89 were monitored simultaneously. A calibration curve and chromatograms are presented in Figures 5.12 and 5.13 respectively.

Oxidized lipid extract (500 μg) was dissolved in chloroform : methanol (1 : 1, 1 mL), and an aliquot was diluted 1 : 10 (v/v) in methanol. To this solution (90 μL) was added the M-PE standard (10 μL, 1 ng/μL in methanol), and the resulting mixture (10 μL) was analyzed by LC-MS/MS using the same HPLC gradient and MS/MS conditions as described above for authentic lysoPE-CEP. The mass transitions, m/z 574.07 → 409.29,

574.07 → 317.98, 574.07 → 255.22, 574.07 → 152.89 were monitored simultaneously.

Chromatograms are shown in Figure 5.11. The absolute amount of lysoCEP-PE was 260 pg in 1 μg lipid extract from UV-promoted oxidized bovine retina. No lysoCEP-PE was detected in lipid extracts from unoxidized bovine retina.

LC/MS/MS analysis and quantification of lysoCEP-PE in bovine retina treated with

PLA2. Lipid extract (1 mg) and PO-PC (1 mg) were suspended in PBS buffer (1 mL, 10

mM, pH 7.4). The mixture was vortexed for 1 min, sonicated for 5 min and passed

through an extruder. CaCl2 (5 mM, 70 μL) in pH 7.4 PBS solution (10 mM, 1 mL) was added. The system was sonicated for 2 min followed by the addition of PLA2 (10,000

193 unit/mL, 10 μL) and then incubated in 37 °C for 5 h. Lipid was then extracted using

Bligh and Dyer method. Lipid extract was dried under a stream of argon. Lipid (400 μg)

was dissolved in methanol (400 μL). To this solution (90 μL) was added the M-PE standard (10 μL, 1 ng/μL in MeOH), and the lipid mixture (10 μL) was analyzed by

LC-MS/MS using the same HPLC gradient and MS/MS conditions as described above for pure lysoPE-CEP. The mass transitions, m/z 574.07 → 409.29, 574.07 → 317.98,

574.07 → 255.22, 574.07 → 152.89 were monitored simultaneously. No peaks expected for lysoCEP-PE were detected.

LC/MS/MS analysis and quantification of lysoCEP-PE in human plasma.

LysoCEP-PE (50 ng in 20 μL methanol) was injected onto a Waters 2790 HPLC system, eluting with 1 mM ammonium acetate in water and using a linear gradient to 100% methanol over 4 minutes, followed by 100% methanol for 9 minutes. Mass spectrometric analysis was performed online using an ESI tandem mass spectrometer in the negative ion multiple reaction monitoring (MRM) mode (cone energy, 45 V; collision energy, 24 eV). The total ion current was measured in the mass range of m/z 200-1000 in the negative ion mode. A 3.77 kilovolt potential was applied to the electrospray capillary. The optimal collision energy to obtain the most intense daughters was determined to be 24 eV.

The following mass transitions, m/z 574.07 → 409.29, 574.07 → 317.98, 574.07 →

255.22, 574.07 → 152.89 were monitored simultaneously. The chromatograms are presented in Figure 5.12.

Lipids were extracted from plasma samples of a normal control and an AMD patient

194 using the Bligh and Dyer method as described above. Lipid extracts (300 μg) from an

AMD plasma sample or (420 μg) from a normal plasma sample were dissolved in methanol (60 μL). Then the M-PE standard (10 μL, 1 ng/μL in methanol) was added, and the resulting mixture (20 μL) was analyzed by LC-MS/MS using the same HPLC gradient and MS/MS conditions as described above for authentic lysoPE-CEP. The mass transitions, m/z 574.07 → 409.29, 574.07 → 317.98, 574.07 → 255.22, 574.07 → 152.89 were monitored simultaneously. Chromatograms of normal and AMD samples are shown in Figures 5.13 and 5.14. There was 1.2 ng lysoCEP-PE in 1 mL plasma from a normal control and 1.3 ng lysoCEP-PE in 1 mL plasma from an AMD patient.

195 5.5. References

1. Fliesler, S. J.; Anderson, R. E., Chemistry and Metabolism of Lipids in the Vertebrate

Retina. Prog. Lipid. Res. 1983, 22, (2), 79-131.

2. Boulton, M.; Rozanowska, M.; Rozanowski, B., J. Photochem. Pathobiol. B: Biology

2001, 64.

3. Young, R. W., The renewal of photoreceptor cell outer segments. J. Cell. Biol. 1967,

33, 61-72.

4. Young, R. W.; and Bok, D., Participation of the retinal pigment epithelium in the rod

outer segment renewal process. J. Cell. Biol. 1969, 42, 392-403.

5. LaVail, M. M., Rod outer segment disk shedding in rat retina: relationship to cyclic

lighting. Science 1976, 194, 1071-1074.

6. Rozanowska, M.; Jarvis-Evans, J.; Korytowski, W.; Boulton, M. E.; Burke, J. M.;

Sarna, T., Blue light-induced reactivity of retinal age pigment. In vitro generation of

oxygen-reactive species. J. Biol. Chem. 1995, 270, (32), 18825-30.

7. Chen, P.; Wiesler, D.; Chmelı´k, J.; Novotny, M., Substituted

2-Hydroxy-1,2-dihydropyrrol-3-ones: Fluorescent Markers Pertaining to Oxidative Stress and Aging. Chem. Res. Toxicol. 1996, 9, 970-979.

8. Kikugawa, K.; Kato, T.; Beppu, M.; Hayasaka, A., Fluorescent and cross-linked

proteins formed by free radical and aldehyde species generated during lipid oxidation.

Adv. Exp. Med. Biol. 1989, 266, 345-356; discussion 357.

9. Gaillard, E. R.; Atherton, S. J.; Eldred, G.; Dillon, J., Photophysical studies on human

196 retinal lipofuscin. Photochem. photobiol. 1995, 61, (5), 448-53.

10. van der Schaft, T. L.; de Bruijn, W. C.; Mooy, C. M.; Ketelaars, D. A.; de Jong, P. T.,

Element analysis of the early stages of age-related macular degeneration. Arch.

Ophthalmol. 1992, 110, (3), 389-94.

11. Liu, J.; Itagaki, Y.; Ben-Shabat, S.; Nakanishi, K.; Sparrow, J. R., The biosynthesis of

A2E, a fluorophore of aging retina, involves the formation of the precursor, A2-PE, in the photoreceptor outer segment membrane. J. Biol. Chem. 2000, 275, (38), 29354-60.

12. Sakai, N.; Decatur, J.; Nakanishi, K., J. Am. Chem. Soc. 1996, 118, 1559-1560.

13. Parish, C. A.; Hashimoto, M.; Nakanishi, K.; Dillon, J.; Sparrow, J., Isolation and one-step preparation of A2E and iso-A2E, fluorophores from human retinal pigment epithelium. Proc. Natl. Acad. Sci. U. S. A. 1998, 95, (25), 14609-13.

14. Sun, H.; Nathans, J., ABCR, the ATP-binding cassette transporter responsible for

Stargardt macular dystrophy, is an efficient target of all-trans-retinal-mediated photooxidative damage in vitro. Implications for retinal disease. J. Biol. Chem. 2001, 276,

(15), 11766-74.

15. Ben-Shabat, S.; Itagaki, Y.; Jockusch, S.; Sparrow, J. R.; Turro, N. J.; Nakanishi, K.,

Formation of a nonaoxirane from A2E, a lipofuscin fluorophore related to macular degeneration, and evidence of singlet oxygen involvement. Angew. Chem. Int. Ed Engl.

2002, 41, (5), 814-7.

16. Sparrow, J. R.; Zhou, J.; Ben-Shabat, S.; Vollmer, H.; Itagaki, Y.; Nakanishi, K.,

Involvement of oxidative mechanisms in blue-light-induced damage to A2E-laden RPE.

197 Invest. Ophthalmol. Vis. Sci. 2002, 43, (4), 1222-7.

17. Jang, Y. P.; Matsuda, H.; Itagaki, Y.; Nakanishi, K.; Sparrow, J. R., Characterization

of peroxy-A2E and furan-A2E photooxidation products and detection in human and

mouse retinal pigment epithelial cell lipofuscin. J. Biol. Chem. 2005, 280, (48), 39732-9.

18. Zhou, J.; Cai, B.; Jang, Y. P.; Pachydaki, S.; Schmidt, A. M.; Sparrow, J. R.,

Mechanisms for the induction of HNE- MDA- and AGE-adducts, RAGE and VEGF in

retinal pigment epithelial cells. Exp. Eye Res. 2005, 80, (4), 567-80.

19. Holz, F. G.; Schutt, F.; Kopitz, J.; Eldred, G. E.; Kruse, F. E.; Volcker, H. E.; Cantz,

M., Inhibition of lysosomal degradative functions in RPE cells by a retinoid component

of lipofuscin. Invest. Ophthalmol. Vis. Sci. 1999, 40, (3), 737-43.

20. Sparrow, J. R.; Cai, B., Blue light-induced apoptosis of A2E-containing RPE:

involvement of caspase-3 and protection by Bcl-2. Invest. Ophthalmol. Vis. Sci. 2001, 42,

(6), 1356-62.

21. Sparrow, J. R.; Nakanishi, K.; Parish, C. A., The lipofuscin fluorophore A2E mediates blue light-induced damage to retinal pigmented epithelial cells. Invest.

Ophthalmol. Vis. Sci. 2000, 41, (7), 1981-9.

22. Schutt, F.; Bergmann, M.; Holz, F. G.; Dithmar, S.; Volcker, H. E.; Kopitz, J.,

Accumulation of A2-E in mitochondrial membranes of cultured RPE cells. Graefes. Arch.

Clin. Exp. Ophthalmol. 2006.

23. Bucala, R.; Makita, Z.; Koschinsky, T.; Cerami, A.; Vlassara, H., Lipid Advanced

Glycosylation - Pathway for Lipid Oxidation in-Vivo. Proc. Natl. Acad. Sci. U. S. A. 1993,

198 90, (14), 6434-6438.

24. Lertsiri, S.; Shiraishi, M.; Miyazawa, T., Identification of deoxy-D-fructosyl

phosphatidylethanolamine as a non-enzymic glycation product of

phosphatidylethanolamine and its occurrence in human blood plasma and red blood cells.

Biosci. Biotech. Biochem. 1998, 62, (5), 893-901.

25. Ravandi, A.; Kuksis, A.; Marai, L.; Myher, J. J., Preparation and Characterization of

Glucosylated Aminoglycerophospholipids. Lipids 1995, 30, (10), 885-891.

26. Ravandi, A.; Kuksis, A.; Marai, L.; Myher, J. J.; Steiner, G.; Lewisa, G.; Kamido, H.,

Isolation and identification of glycated aminophospholipids from red cells and plasma of

diabetic blood. FEBS Lett. 1996, 381, (1-2), 77-81.

27. Requena, J. R.; Ahmed, M. U.; Fountain, C. W.; Degenhardt, T. P.; Reddy, S.; Perez,

C.; Lyons, T. J.; Jenkins, A. J.; Baynes, J. W.; Thorpe, S. R., Carboxymethylethanolamine, a biomarker of phospholipid modification during the maillard reaction in vivo. J. Biol.

Chem. 1997, 272, (28), 17473-17479.

28. Tappel, A. L., Measurement of and protection from in vivo lipid peroxidation. In:

Pryor WA, ed,. Free Radic. Biol. 1980, vol 4. New York: Academic Press, 2-47.

29. Tsuji, K.; Kawai, Y.; Kato, Y.; Osawa, T., Formation of N-(hexanoyl)ethanolamine, a

novel phosphatidylethanolamine adduct, during the oxidation of erythrocyte membrane and low-density lipoprotein. Biochem. Biophys. Res. Commun. 2003, 306, (3), 706-711.

30. Guichardant, M.; Bernoud-Hubac, N.; Chantegrel, B.; Deshayes, C.; Lagarde, M.,

Aldehydes from n-6 fatty acid peroxidation. Effects on aminophospholipids.

199 Prostaglandins Leukot. Essent. Fatty Acids 2002, 67, (2-3), 147-149.

31. Zamora, R.; Hidalgo, F. J., Phosphatidylethanolamine modification by oxidative

stress product 4, 5 (E)-epoxy-2 (E)-heptenal. Chem. Res. Toxicol. 2003, 16, (12),

1632-1641.

32. Bacot, S.; Bernoud-Hubac, N.; Baddas, N.; Chantegrel, B.; Deshayes, C.; Doutheau,

A.; Lagarde, M.; Guichardant, M., Covalent binding of hydroxy-alkenals 4-HDDE,

4-HHE, and 4-HNE to ethanolamine phospholipid subclasses. J. Lip. Res. 2003, 44, (5),

917-926.

33. Bernoud-Hubac, N.; Fay, L. B.; Armarnath, V.; Guichardant, M.; Bacot, S.; Davies, S.

S.; Jackson Roberts II, L.; Lagarde, M., Covalent binding of isoketals to ethanolamine

phosphalipids. Free Radic. Biol. Med. 2004, 37, 1604-1611.

34. Zieseniss, S.; Zahler, S.; Muller, I.; Hermetter, A.; Engelmann, B., Modified phosphatidylethanolamine as the active component of oxidized low density lipoprotein promoting platelet prothrombinase activity. J. Biol. Chem. 2001, 276, (23), 19828-19835.

35. Heller, J. I.; Crowley, J. R.; Hazen, S. L.; Salvay, D. M.; Wagner, P.; Pennathur, S.;

Heinecke, J. W., p-Hydroxyphenylacetaldehyde, an aldehyde generated by myeloperoxidase, modifies phospholipid amino groups of low density lipoprotein in human atherosclerotic intima. J. Biol. Chem. 2000, 275, (14), 9957-9962.

36. Oak, J. H.; Nakagawa, K.; Miyazawa, T., Synthetically prepared Amadori-glycated phosphatidylethanolamine call trigger lipid peroxidation via free radical reactions. FEBS

Lett. 2000, 481, (1), 26-30.

200 37. Gu, X.; Sun, M.; Gugiu, B.; Hazen, S.; Crabb, J. W.; Salomon, R. G., Oxidatively

truncated docosahexaenoate phospholipids: total synthesis, generation, and Peptide

adduction chemistry. J. Org. Chem. 2003, 68, (10), 3749-61.

38. Crabb, J. W.; Miyagi, M.; Gu, X.; Shadrach, K.; West, K. A.; Sakaguchi, H.; Kamei,

M.; Hasan, A.; Yan, L.; Rayborn, M. E.; Salomon, R. G.; Hollyfield, J. G., Drusen proteome analysis: an approach to the etiology of age-related macular degeneration. Proc.

Natl. Acad. Sci. U. S. A. 2002, 99, (23), 14682-7.

39. Ebrahem, Q.; Renganathan, K.; Sears, J.; Vasanji, A.; Gu, X.; Lu, L.; Salomon, R. G.;

Crabb, J. W.; Anand-Apte, B., Carboxyethylpyrrole oxidative protein modifications stimulate neovascularization: Implications for age-related macular degeneration. Proc.

Natl. Acad. Sci. U. S. A. 2006, 103, (36), 13480-4.

40. Bicknell, I. R.; Darrow, R.; Barsalou, L.; Fliesler, S. J.; Organisciak, D. T., Mol. Vis.

2002, 8, 333-340.

41. Bligh, E.; and Dyer, W., A rapid method of total lipid extraction and purification. Can.

J. Biochem. Physiol. 1956, 37, 911-917.

42. Dennis, E. A., Diversity of group types, regulation, and function of phospholipase A2.

J. Biol. Chem. 1994, 269, 13057-13060.

43. Murthi, K. K.; Friedman, L. R.; Oleinick, N. L.; Salomon, R. G., Formation of

DNA-protein cross-links in mammalian cells by levuglandin E2. Biochemistry 1993, 32,

4090-4097.

44. Jain, S. K., The accumulation of malonyldialdehyde, a product of fatty acid

201 peroxidation, can disturb aminophospholipid organization in the membrane bilayer of human erythrocytes. J. Biol. Chem. 1984, 259, (6), 3391-4.

45. Tenchov, B.; Vescio, E. M.; Sprott, G. D.; Zeidel, M. L.; Mathai, J. C., Salt tolerance of archaeal extremely halophilic lipid membranes. J. Biol. Chem. 2006, 281, (15),

10016-23.

202

Chapter 6

Pilot Studies towards Identification of Levuglandin Modified Proteins in

Cornea

203 6.1. Introduction

6.1.1. LPS and inflammation

Inflammation involves the interaction of multiple cell types at the target site releasing

eicosanoids (prostaglandins, prostacyclins, thromboxanes and leukotrienes), proteolytic

enzymes, cytokines, and other inflammatory mediators.1 Insults to the endothelial layer of

the blood vessels can stimulate the production of leukocyte chemo-attractant molecules

and the expression of adhesion molecules that bind to the monocytes and make them

adhere to the endothelial layer through integrins. The activation of chemotactic factors,

e.g., monocyte chemoattractant protein-1 (MCP-1), usually in response to inflammatory

cytokines such as interleukin-1 (IL-1), interleukin-4 (IL-4), tumor necrosis factor α

(TNF-α), and interferon-γ (IFN-γ), recruits more phagocytic and immune cells to the site

of inflammation.2-5 It is well established that lipopolysaccharide (LPS) stimulates a

variety of inflammatory responses including activation of the alternate complement

cascade and the production of cytokines.6 LPS, a structural element of Gram-negative

bacterial cell wall consisting of lipids and polymers of carbohydrates, serves as a ligand

for receptors that can activate proinflammatory cytokines in many cell types.6-8 Acute

exposure to LPS in serum concentrations as low at 1 ng/mg could cause death.9 Released cytokines, such as tumor necrosis factor and interleukin-1, can activate endothelial cells

to upregulate receptors for various immune cells.10, 11

6.1.2. The cyclooxygenase and isoprostane pathways. Cyclooxygenase (COX), also

known as prostaglandin endoperoxide (PGH2) synthase, is the first enzyme of the

204 pathway by which arachidonic acid (AA) is highly stereoselectively cyclooxygenated to a

12 single, enantiomerically pure prostaglandin endoperoxide PGH2. The existence of two

cyclooxygenases in human endothelial cells has been proven. COX-1 is considered a

constitutive enzyme, being found in most mammalian cells. More recently it has been

shown to be upregulated in several carcinomas and to have a central role in tumorigenesis.

COX-2 is undetectable in most normal tissues and is induced in a number of cell types

including endothelial cells,13 neutrophils,13, 14 macrophages15, 16 and monocytes16, 17 by a

variety of inflammatory mediators, such as IL-1, IL-2 and LPS1, 18-20 at sites of

inflammation.

PGH2, which is generated enzymatically through the action of COX from arachidonic

acid (AA), is a branch point in the biosynthesis of numerous hormone-like mediators

21, 22 (PGD2, PGE2, PGI2, PGF2, and thromboxane TXA2) of normal physiological

processes (Figure 6.1). Non-enzymatical rearrangement of PGH2 in protic solvents

produces two levulinaldehyde derivatives with prostanoid side chains which were named

23 levuglandin (LG) D2 and E2. In addition, free radical-induced cyclooxygenation of AA

and its phospholipid esters through the isoprostane pathway generates isomers of PGs

which were coined isoprostanes (isoPs).24, 25 Non-enzymatic rearrangement of isoP endoperoxides gives rise to both stereoisomers and structural isomers of levuglandins,

26 which were defined as isoLGs and iso[n]LGs respectively. LGE2, one of the stereoisomers designated collectively as isoLGE2, is a product of both the COX and

isoprostane pathways whereas iso[4]LGE2 derives only from isoprostane pathway

205 (Scheme 6.1).

Figure 6.1. Eicosanoid synthesis.

(CH2)3COOPC (CH ) COOH Phospholipase A2 2 3

C5H11 C5H11 AA-PC AA

Free radical induced Cyclooxygenase

(CH2)3COOPC O O (CH2)3COOH O O C5H11 C5H11 OH OH

isoPGH2-PC PGH2

O O (CH2)3COOPC (CH2)3COOH

OHC C5H11 OHC C H 5 11 OH OH Levuglandin E2 Iso[4]levuglandin E2-PC

Scheme 6.1. Representative pathways showing enzyme and free radical induced generation of LGE2 and iso[4]LGE2 from AA-PC.

206 6.1.3. LGE2 and iso[4]LGE2 bind avidly with proteins. LGE2 and iso[4]LGE2 have exceptional reactivity to biological nucleophiles such as proteins and DNA.27-29 It was

proposed that levuglandins are the reactive electrophiles responsible for much (if not all)

of the COX-dependent binding of AA metabolites.30 Upon incubation of bovine serum

3 albumin (BSA) with an excess of 5,6- H-LGE2, 10 molecules of LGE2 irreversibly bind

30 to BSA within 1 min. LGE2 and iso[4]LGE2 protein adducts were found in human

plasma by immunoassays using antibodies raised against corresponding LGE2 and

31 iso[4]LGE2 keyhole limpet hemocyanin (KLH) adducts. More importantly, the levels of

both LGE2 and iso[4]LGE2 protein adduct immunoreactivity were significantly elevated

in plasma from end-stage renal disease (RD) and atherosclerosis (AS) than in plasma from healthy controls.32 These clinical findings provide unambiguous evidence that lipid

oxidation is correlated to pathological disease.

6.1.4. Pyridoxamine, a potent trap that prevents protein modification by reactive

electrophiles. In view of the damage caused by protein modification by products of lipid

oxidation, many approaches have been explored to protect against the assault of lipid

oxidation products on amino groups of proteins.33 Pyridoxamine (vitamine B6), an

essentially nontoxic amine, was first proposed by Hudson and colleagues as an inhibitor

of the formation of advanced glycoxidation end products (AGEs).34, 35 Recently,

pyridoxamine was documented to be an extremely potent scavenger of 1,4-dicarbonyl

compounds. Pyridoxamine reacted with 1,4-dicarbonyls at rates 2300 times greater than

to α–N-AcLys in the formation of pyrroles and it strongly reduced adduction to lysine by

207 a γ-ketoaldehyde when present at equimolar concentrations to α–N-AcLys in vitro.36 The

acidic phenol group on pyridoxamine was proposed to be responsible for its extraordinary

reactivity. This hydroxyl protonates the carbonyl in a hemiaminal intermediate and holds

it into a conformation that favors intramolecular attack of the amine, facilitating

cyclization to a pyrrolidine diol (Scheme 6.2).36 In animal model studies, pyridoxamine,

administrated in drinking water at 1 g/l, inhibited AGE formation and retarded the

development of early renal disease. It also reduced the development of acellular

capillaries which is one of the earliest hallmarks of diabetic retinopathy in

streptozotocin-induced diabetic rats.37-40 All these experiments encourage us to examine

the ability of pyridoxamine to inhibit the accumulation of LG modifications.

In this chapter, a pilot study was constructed on the LPS induced formation of LGE2 and iso[4]LGE2 protein adducts in a mouse cornea model. Further studies are planned to test the ability of pyridoxamine to prevent LPS-promoted haze development using mouse models.

R2 R R H O HO 2 2 H H OH R2 O N O O NH2 O N N H R1 + OH R1 N OH N HO R1 O OH N N OH OH R1 pyridoxamine hemiaminal pyrrolidine diol pyrrole

Scheme 6.2. Possible mechanism of pyrrole formation between pyridoxamine and

1,4-dicarbonyls.

208 6.2. Results and Discussion

Cornea of C57BL/6 mouse was treated in vivo with ultrapure LPS (2 μg) in water (2

μL) or PBS (2 μL) by intrastromal injection. Alternatively, the cornea was scratched three times and then LPS in water (2 μL) or PBS alone (2 μL) was dropped onto the scratches.

After 24 h, the cornea was excised. To localize the LGE2 and iso[4]LGE2 modified

proteins in the cornea and to determine the effect of LPS on the generation of LG- or

isoLG-protein adducts, corneal sections were stained with the corresponding antibodies.

The slides were then incubated with a fluorescent second antibody and analyzed with a

fluorescence microscope. The results are presented in Figure 6.2. The green fluorescence

represents the staining of LGE2-protein or iso[4]LGE2-protein adducts, while the blue

represents nuclear staining by 4,6-diamidino- -2-phenylindole (DAPI). The LPS-injected

cornea showed visibly elevated green fluorescence of both LGE2-protein and

iso[4]LGE2-protein adducts compared to the PBS-injected cornea. However, cornea

scratched and treated with LPS did not seem to show expected differences compared with

PBS treatment.

In the eye, corneal inflammation caused by microbial keratitis or bacterial products, e.

g., LPS, leads to a significant loss of vision.41 The translucent nature of the cornea allows in vivo visualization of inflammatory events in a noninvasive manner.42 Intrastromal

injection of LPS has been reported to induce COX-2 expression.43-45 As mentioned above,

COX-2 catalyzes the synthesis of PGH2, which undergoes non-enzymatic rearrangement

to give rise to LGE2. Iso[4]LGE2 is a unique product from free-radical induced

209 Untreated cyclooxygenation.A The resultsB show that LPS injection C

D E F PBS LPS

G H I

K J L Untreated

M N O PBS

P Q R LPS

Preimmune LGE2 Iso[4]LGE2

Figure 6.2. Cornea of C57BL/6 mouse was treated in vivo with LPS or PBS for 24 h by injection or scratch. Sections (5 μm) of cornea were stained with LGE2 and iso[4]LGE2 pAb. A, B, C, J, K, L – Untreated; D, E, F – PBS injection; G, H, I – LPS injection; M, N, O – PBS treated (scratch); P, Q, R – LPS treated (scratch).

Panels treated with preimmune, LGE2 and iso[4]LGE2 pAbs are indicated in the Figure.

210 increased both LGE2-protein and iso[4]LGE2-protein adducts. The formation of

iso[4]LGE2-protein adducts confirms that free radical mediated lipid oxidation is promoted by LPS. However, the formation of LGE2-protein adducts does not show that

LPS induces COX pathway because these adducts are also expected to be generated

through free radical-induced lipid oxidation, i. e., the isoprostane pathway.

Further experiments in continuation of this pilot project should be focused on

examining the efficacy of pyridoxamine for scavenging endogenous γ-ketoaldehydes and

retarding the accumulation of LGE2-protein or iso[4]LGE2-protein modifications, and

thereby preventing haze development in keratitis.

211 6.3. Experimental Procedures.

General methods. The following commercially available materials were used as

received: fetal calf serum (FCS, Invitrogen, Carlsbad, CA); phosphate buffered saline

(PBS, Sigma, St. Louis, MO); goat anti-rabbit IgG conjugated with Alexa 1000 and

Vectashield mounted with media containing 4,6-diamidino-2-phenylindole (DAPI) were

from Vector Laboratory Inc, Burlingam, CA; Anti-LGE2-KLH and anti-iso[4]LGE2-KLH polyclonal antibodies were purified as described previously.26

Immunohistochemical analysis. Eyes were enucleated 24 hours after intrastromal

injection or scratch, and snap frozen in optimum temperature cutting compound

(Tissue-Tek; Miles Scientific, Napierville, IL). Five-micrometer frozen sections of the

central cornea were cut and fixed on slides for immunohistochemical analysis. The slides

were fixed by submersion 4% paraformaldehyde solution for 30 min, rinsed with PBS

and then blocked with 2% fetal calf serum (FCS) (200 μL/slide) for 30 min at room

temperature. Subsequently, the slides were incubated with appropriate first antibody

solutions (200 μL/slide) for 2 h at room temperature. The antibody solution was prepared

by adding 25 μL of concentrated antibody (1.94 mg/mL polyclonal rabbit anti-LGE2 or

1.14 mg/mL polyclonal rabbit anti-iso[4]LGE2) into 2.5 mL 1% FCS in PBS containing

3% dry milk. The slides were then washed with 1% FCS in PBS (3 x 5 min) and

subsequently incubated with goat anti-rabbit IgG conjugated with Alexa 1000 (Vector

Laboratory Inc, Burlingam, CA) (10 ng/slide) for 1 h at room temperature. Slides were

washed with 1% FCS in PBS three times and with PBS two times. Slides were dried

212 carefully with kimwipes, sealed with Vectashield, mounted with media containing DAPI

(blue) (Vector Laboratory Inc, Burlingam, CA), and analyzed with a Nikon EFD-3 fluorescence microscope attached to a CCD camera.

213 6.4. References

1. Fu, J. Y.; Masferrer, J. L.; Seibert, K.; Raz, A.; Needleman, P., The induction and

suppression of prostaglandin H2 synthase (cyclooxygenase) in human monocytes. J. Biol.

Chem. 1990, 265, (28), 16737-40.

2. Zhang, Y.; Ramos, B. F.; Jakschik, B. A., Neutrophil recruitment by tumor necrosis

factor from mast cells in immune complex peritonitis. Science 1992, 258, (5090), 1957-9.

3. Rollins, B. J.; Pober, J. S., Interleukin-4 induces the synthesis and secretion of

MCP-1/JE by human endothelial cells. Am. J. Pathol. 1991, 138, (6), 1315-9.

4. Rollins, B. J.; Yoshimura, T.; Leonard, E. J.; Pober, J. S., Cytokine-activated human endothelial cells synthesize and secrete a monocyte chemoattractant, MCP-1/JE.

Am. J. Pathol. 1990, 136, (6), 1229-33.

5. Peters, W.; Charo, I. F., Involvement of chemokine receptor 2 and its ligand, monocyte chemoattractant protein-1, in the development of atherosclerosis: lessons from knockout mice. Curr. Opin. Lipidol. 2001, 12, (2), 175-80.

6. Schultz, C. L.; Morck, D. W.; McKay, S. G.; Olson, M. E.; Buret, A.,

Lipopolysaccharide induced acute red eye and corneal ulcers. Exp. Eye Res. 1997, 64, (1),

3-9.

7. Chetty, C.; Klapper, D. G.; Schwab, J. H., Soluble peptidoglycan-polysaccharide fragments of the bacterial cell wall induce acute inflammation. Infect Immun. 1982, 38,

(3), 1010-9.

8. Esser, R. E.; Anderle, S. K.; Chetty, C.; Stimpson, S. A.; Cromartie, W. J.; Schwab,

214 J. H., Comparison of inflammatory reactions induced by intraarticular injection of bacterial cell wall polymers. Am. J. Pathol. 1986, 122, (2), 323-34.

9. Rustici, A.; Velucchi, M.; Faggioni, R.; Sironi, M.; Ghezzi, P.; Quataert, S.; Green,

B.; Porro, M., Molecular mapping and detoxification of the lipid A binding site by synthetic peptides. Science 1993, 259, (5093), 361-5.

10. Dinarello, C. A.; Mier, J. W., Interleukins. Annu Rev Med 1986, 37, 173-8.

11. Dinarello, C. A.; Conti, P.; Mier, J. W., Effects of human interleukin-1 on natural killer cell activity: is fever a host defense mechanism for tumor killing? Yale J. Biol. Med.

1986, 59, (2), 97-106.

12. Salomon, R. G., Distinguishing levuglandins produced through the cyclooxygenase and isoprostane pathways. Chem. Phys. Lipids 2005, 134, (1), 1-20.

13. Bottoms, G. D.; Johnson, M. A.; Lamar, C. H.; Fessler, J. F.; Turek, J. J.,

Endotoxin-induced eicosanoid production by equine vascular endothelial cells and neutrophils. Circ. Shock 1985, 15, (3), 155-62.

14. Doerfler, M. E.; Danner, R. L.; Shelhamer, J. H.; Parrillo, J. E., Bacterial lipopolysaccharides prime human neutrophils for enhanced production of leukotriene B4.

J. Clin. Invest. 1989, 83, (3), 970-7.

15. Aderem, A. A.; Cohen, D. S.; Wright, S. D.; Cohn, Z. A., Bacterial lipopolysaccharides prime macrophages for enhanced release of arachidonic acid metabolites. J. Exp. Med. 1986, 164, (1), 165-79.

16. Kurland, J. I.; Bockman, R., Prostaglandin E production by human blood

215 monocytes and mouse peritoneal macrophages. J. Exp. Med. 1978, 147, (3), 952-7.

17. Nichols, F. C.; Schenkein, H. A.; Rutherford, R. B., Prostaglandin E2, prostaglandin E1 and thromboxane B2 release from human monocytes treated with C3b or bacterial lipopolysaccharide. Biochim. Biophys. Acta. 1987, 927, (2), 149-57.

18. Maier, J. A.; Hla, T.; Maciag, T., Cyclooxygenase is an immediate-early gene induced by interleukin-1 in human endothelial cells. J. Biol. Chem. 1990, 265, (19),

10805-8.

19. Raz, A.; Wyche, A.; Needleman, P., Temporal and pharmacological division of fibroblast cyclooxygenase expression into transcriptional and translational phases. Proc.

Natl. Acad. Sci. U. S. A. 1989, 86, (5), 1657-61.

20. Raz, A.; Wyche, A.; Siegel, N.; Needleman, P., Regulation of fibroblast cyclooxygenase synthesis by interleukin-1. J. Biol. Chem. 1988, 263, (6), 3022-8.

21. Hamberg, M.; Samuelsson, B., Detection and isolation of an endoperoxide intermediate in prostaglandin biosynthesis. Proc. Natl. Acad. Sci. U. S. A. 1973, 70, (3),

899-903.

22. Nugteren, D. H.; Hazelhof, E., Isolation and properties of intermediates in prostaglandin biosynthesis. Biochim. Biophys. Acta. 1973, 326, (3), 448-61.

23. Salomon, R. G. M., D. B.; Zagorski, M. G.; and Coughlin, D.J., Prostaglandin

Endoperoxides. 14. Solvent-induced fragmentation of prostaglandin endoperoxides. New aldehyde products from PGH2 and a novel intramolecular 1,2-hydride shift during endoperoxide fragmentation in aqueous solution. J. Am. Chem. Soc. 1984, 106,

216 6049-6060.

24. Nugteren, D. H. V., H.; Van Dorp, D. A., Nonenzymic conversion of all-cis

8,11,14-eicosatrienoic acid into prostaglandin E1. Rec. Trav. Chim. 1967, 86, 1237-1245.

25. Morrow, J. D.; Hill, K. E.; Burk, R. F.; Nammour, T. M.; Badr, K. F.; Roberts, L.

J., 2nd, A series of prostaglandin F2-like compounds are produced in vivo in humans by a non-cyclooxygenase, free radical-catalyzed mechanism. Proc. Natl. Acad. Sci. U. S. A.

1990, 87, (23), 9383-7.

26. Salomon, R. G.; Subbanagounder, G.; Singh, U.; O'Neil, J.; Hoff, H. F., Oxidation of low-density lipoproteins produces levuglandin-protein adducts. Chem. Res. Toxicol.

1997, 10, (7), 750-9.

27. Boutaud, O.; Brame, C. J.; Salomon, R. G.; Roberts, L. J., 2nd; Oates, J. A.,

Characterization of the lysyl adducts formed from prostaglandin H2 via the levuglandin pathway. Biochemistry 1999, 38, (29), 9389-96.

28. Murthi, K. K.; Friedman, L. R.; Oleinick, N. L.; Salomon, R. G., Formation of

DNA-protein cross-links in mammalian cells by levuglandin E2. Biochemistry 1993, 32,

(15), 4090-7.

29. Salomon, R. G., Levuglandins and isolevuglandins: stealthy toxins of oxidative injury. Antioxid. Redox. Signal 2005, 7, (1-2), 185-201.

30. Salomon, R. G.; Jirousek, M. R.; Ghosh, S.; Sharma, R. B., Prostaglandin endoperoxides 21. Covalent binding of levuglandin E2 with proteins. Prostaglandins

1987, 34, (5), 643-56.

217 31. Salomon, R. G.; Batyreva, E.; Kaur, K.; Sprecher, D. L.; Schreiber, M. J.; Crabb,

J. W.; Penn, M. S.; DiCorletoe, A. M.; Hazen, S. L.; Podrez, E. A., Isolevuglandin-protein adducts in humans: products of free radical-induced lipid oxidation through the isoprostane pathway. Biochim. Biophys. Acta 2000, 1485, (2-3), 225-35.

32. Salomon, R. G.; Sha, W.; Brame, C.; Kaur, K.; Subbanagounder, G.; O'Neil, J.;

Hoff, H. F.; Roberts, L. J., 2nd, Protein adducts of iso[4]levuglandin E2, a product of the isoprostane pathway, in oxidized low density lipoprotein. J. Biol. Chem. 1999, 274, (29),

20271-80.

33. Davies, S. S.; Brantley, E. J.; Voziyan, P. A.; Amarnath, V.; Zagol-Ikapitte, I.;

Boutaud, O.; Hudson, B. G.; Oates, J. A.; Ii, L. J., Pyridoxamine Analogues Scavenge

Lipid-Derived gamma-Ketoaldehydes and Protect against H(2)O(2)-Mediated

Cytotoxicity. Biochemistry 2006, 45, (51), 15756-15767.

34. Khalifah, R. G.; Todd, P.; Booth, A. A.; Yang, S. X.; Mott, J. D.; Hudson, B. G.,

Kinetics of nonenzymatic glycation of ribonuclease A leading to advanced glycation end products. Paradoxical inhibition by ribose leads to facile isolation of protein intermediate for rapid post-Amadori studies. Biochemistry 1996, 35, (15), 4645-54.

35. Booth, A. A.; Khalifah, R. G.; Todd, P.; Hudson, B. G., In vitro kinetic studies of formation of antigenic advanced glycation end products (AGEs). Novel inhibition of post-Amadori glycation pathways. J. Biol. Chem. 1997, 272, (9), 5430-7.

36. Amarnath, V.; Amarnath, K.; Amarnath, K.; Davies, S.; Roberts, L. J., 2nd,

Pyridoxamine: an extremely potent scavenger of 1,4-dicarbonyls. Chem. Res. Toxicol.

218 2004, 17, (3), 410-5.

37. Alderson, N. L.; Chachich, M. E.; Frizzell, N.; Canning, P.; Metz, T. O.;

Januszewski, A. S.; Youssef, N. N.; Stitt, A. W.; Baynes, J. W.; Thorpe, S. R., Effect of

antioxidants and ACE inhibition on chemical modification of proteins and progression of

nephropathy in the streptozotocin diabetic rat. Diabetologia. 2004, 47, 1385-1395.

38. Alderson, N. L.; Chachich, M. E.; Youssef, N. N.; Beattie, R. J.; Nachtigal, M.;

Thorpe, S. R.; Baynes, J. W., The AGE inhibitor pyridoxamine inhibits lipemia and development of renal and vascular disease in Zucker obese rats. Kidney Int. 2003, 63, (6),

2123-33.

39. Degenhardt, T. P.; Alderson, N. L.; Arrington, D. D.; Beattie, R. J.; Basgen, J. M.;

Steffes, M. W.; Thorpe, S. R.; Baynes, J. W.; , Pyridoxamine inhibits early renal disease

and dyslipidemia in the streptozotocin-diabetic rat. Kidney Int. 2002, 61, 939-950.

40. Stitt, A.; Gardiner, T. A.; Alderson, N. L.; Canning, P. F., N.; ; Duffy, N.; Boyle,

C.; Januszewski, A. S.; Chachich, M.; Baynes, J. W.; Thorpe, S. R., The AGE inhibitor pyridoxamine inhibits development of retinopathy in experimental diabetes. Diabetes

2002, 51, 2826-2832.

41. Lyczak, J. B.; Cannon, C. L.; Pier, G. B., Establishment of Pseudomonas aeruginosa infection: lessons from a versatile opportunist. Microbes and infection /

Institut. Pasteur. 2000, 2, (9), 1051-60.

42. Carlson, E. C.; Drazba, J.; Yang, X.; Perez, V. L., Visualization and characterization of inflammatory cell recruitment and migration through the corneal

219 stroma in endotoxin-induced keratitis. Invest. Ophthalmol. Vis. Sci. 2006, 47, (1), 241-8.

43. Peng, T.; Lu, X.; Feng, Q., NADH oxidase signaling induces cyclooxygenase-2 expression during lipopolysaccharide stimulation in cardiomyocytes. FASEB J. 2005, 19,

(2), 293-5.

44. Ristimaki, A.; Garfinkel, S.; Wessendorf, J.; Maciag, T.; Hla, T., Induction of cyclooxygenase-2 by interleukin-1 alpha. Evidence for post-transcriptional regulation. J.

Biol. Chem. 1994, 269, (16), 11769-75.

45. Hla, T.; Neilson, K., Human cyclooxygenase-2 cDNA. Proc. Natl. Acad. Sci. U. S.

A. 1992, 89, (16), 7384-8.

220

Chapter 7

Syntheses of Isosteric Pyrazole Derivatives

221 7.1. Background

Numerous biological processes are proposed to be mediated through pyrrole formation

on biological molecules, and pyrrole adducts on protein and DNA have been associated

with pathological diseases.1-4 Humans or experimental animals can develop a distal

neuropathy, which is an accumulation of neurofilaments in large myelinated axons

(myelin is an insulating lipid layer formed around nerves), or even lose distal sensory and

motor functions when exposed to hexane. Pyrrole formation, which stems from the

hexane oxidation product 2,5-hexanedione that reacts with lysyl ε-amino groups of

proteins, is believed to be the first step in the induction of neuropathy.3, 4 Peroxidation

products from fatty acids also are known to form pyrroles upon reaction with lysyl

ε-amino groups. For example, 4-hydroxy-2-nonenal (HNE), which has been touted as the

“most cytotoxic aldehyde”5, 6 released during peroxidation of linoleate and arachidonate

fatty esters, has been confirmed to adversely modify proteins and irreversibly generate a

2-pentylpyrrole adduct as well as Michael and Shiff-base adducts.7-10 This HNE-derived

pyrrole has been called an example of an “advanced lipid peroxidation end product”

(ALPE) based on the multistep nature of the condensation which leads to a pyrrole.7

Levuglandin E2 (LGE2), a nonenzymatic rearrangement seco prostanoid product derived from the prostaglandin endoperoxide PGH2 during metabolism of arachidonic acid (AA),

covalently binds to proteins and generates pyrrole derivatives as major primary

11-16 products. Both 2-pentylpyrrole and LGE2 protein adducts can be detected in human

plasma, and are more prevalent in plasma from patients with renal disease or

222 atherosclerosis than in plasma from normal controls,17, 18 making them indelible markers of oxidative injury associated degenerative diseases.

The pyrrole ring is an electron-rich heterocycle, and its derivatives readily undergo

oxidation under photochemical and electrochemical conditions, or in the presence of

molecular oxygen.19, 20 The oxidations are normally sophisticated and generally result in

various products in different distribution.1, 21-23 It has been established that proteins carrying pyrrole rings tend to covalently cross-link under oxidizing conditions. Either

pyrrole-pyrrole dimerization24 or reaction of a putative electrophilic autoxidized pyrrole

moiety with nucleophilic protein side chains such as lysyl ε-amino groups or cysteine

thiols are possible contributions to cross-linking under physiological conditions (Figure

7.1).1, 25, 26 The limited stability of pyrroles detracts from their utility as standards in

immunosorbent assays. When the pyrroles are used as standards, they must be freshly

prepared. For example, HNE-derived 2-pentylpyrroles degrade over a period of one year.

Since each batch contains different amounts of pyrroles and there is always the

possibility that various pyrrole-Nu crosslinks exist simultaneously, it is not reliable to

compare levels of pyrrole determined with the use of different batches of standard.

Therefore, we opted for a more reliable approach that exploits the high crossreactivity anticipated between pyrroles and their stable pyrazole isosteres. The pyrazoles are

electron deficient, and consequently, are unlikely to undergo electrophilic aromatic substitution or oxidation.27

223 R2 R 2 protein N R2 X protein O2 N protein R1 + R2 CH2 N protein R1 HX protein R1 CH3 N protein CH R1 3 X = N, S

Figure 7.1. Pyrroles cross-link with proteins.

Since 2-pentylpyrrole generated by condensation of HNE with the ε–amino groups of

protein lysyl residues are appended to the protein by an n-alkyl tether to the pyrrole ring nitrogen, an isosteric pyrazole hapten was designed that would be linked to a carrier protein by an n-alkyl tether to the 1-position of the pyrazole ring. An aldehyde group was chosen to allow coupling by reductive alkylation with peptides or proteins.

In this chapter, the synthesis of pyrazole derivatives, that are analogues of

2-pentylpyrrole, is reported. These are potential standards applicable in immunosorbent assays.

224 7.2. Results and Discussions

7.2.1. Synthesis of a 1,3-dione. Claisen condensation provides a general route to

1,3-dicarbonyl compounds.28-30 An enol 7.1 was obtained by Claisen condensation, preferentially at the methyl position of 2-heptanone, with diethyl oxalate in 64 % yield

(Scheme 7.1). We found no evidence for condensation at the methylene position since

TLC of the crude product showed only one major spot, our target product. Acidification of this sodium salt generated a dione that exists mainly as the enol 7.1.

O 1, Na, Ethanol O OH 2, HCl + (COOC H ) C H C CO Et C5H11 CH3 2 5 2 5 11 2 64% H 7.1

CO2Et 1, I-(CH2)4CH=CH2 (7.3), COOH NH2-NH2H2O NaH, EtOH, HOAc N N C H 2, NaOH, EtOH 5 11 N C5H11 N 84% H 50% (CH2)4CH=CH2 7.2 7.4

170 oC, 9hr N C5H11 N 45% (CH2)4CH=CH2 7.5 acetone, reflux Cl-(CH2)4CH=CH2 + NaI I-(CH2)4CH=CH2 73% 7.3

Scheme 7.1. Synthesis of 1-alkyl-5-pentylpyrazole through a decarboxylation route.

7.2.2. Synthesis and structure identification of 1-alkyl-5-pentylpyrazole-3-carboxylic acid. Condensation of hydrazine with 1,3-dicarbonyl compounds provides an expeditious construction of pyrazoles.31, 32 An N-substituted pyrazole carboxylic acid 7.4 was

225 prepared from the pyrazole ester 7.2 and iodoalkene 7.3 followed by hydrolysis of the

ester with alcoholic NaOH.

Heteronuclear multiple quantum coherence (HMQC) and heteronuclear multiple bond

coherence (HMBC) were used to identify the pyrazole structure. Notably, in the HMBC

α β γ spectrum, H , Η and H all correlate with the same C*. This confirmed that this product

is the 3-carboxyl-5-pentylpyrazole and not the 3-pentyl-5-carboxylpyrazole in which

β γ H and H would not correlate with the same C* (Figure 7.2). The HMQC and HMBC

data are listed in Table 7.1, and the spectra are displayed in Appendix Figures S59 and

S60.

α γ α H H COOH H γ CH C4H9 H * C N *C N CH N HOOC N C4H9 βH CH βH CH (CH2)3CH=CH2 (CH2)3CH=CH2 3-carboxyl-5-pentyl pyrazole 3-pentyl-5-carboxyl pyrazole Figure 7.2. Structural description of C – H HMBC coupling for both 3-carboxyl-5-pentyl pyrazole and 3-pentyl-5-carboxyl pyrazole.

Table 7.1. HMQC and HMBC data for 7.4 (in parts per million).a

HMQC HMBC α α β γ 6.63 (H )↔ 107.37 144.68 (C*)↔ 6.63 (H ), 4.11 (H ), 2.59 (H ) 4.99 ↔ 114.89 141.69 ↔ 6.63 β 4.11 (H )↔ 49.52 137.98 ↔ 2.10 γ 2.59 (H )↔ 25.70 114.89 ↔ 2.10 0.92 ↔ 13.85 107.37 ↔ 2.59

a The symbol ↔ denotes the C-H correlation; solvent: CDCl3.

226 7.2.3. Decarboxylation of a pyrazole carboxylic acid.33 The melting point of pyrazole

carboxylic acid is 145 °C. Melting point was measured on a Thomas Hoover Unimelt

capillary melting point apparatus and is uncorrected. Copper chromite catalyzes decarboxylation of indole 2-carboxylic acid.34 We heated the acid 7.4 with copper

chromite at 200 - 250 °C in a vacuum, but failed to obtain the decarboxylation product.

Barton decarboxylation35, 36 is a potential route from 7.4 to 7.5. However, this route is

lengthy and involves noxious reagents, and was therefore not pursued. It was reported

that heating such pyrazole carboxylic acids at a temperature a little bit higher than their

melting points causes decarboxylation.31 This thermal decarboxylation proved to be effective when the acid 7.4 was heated at 170 °C for 9 h with no catalyst or solvent.

Although the high temperature resulted in a dark, tar-like residue, the desired product 7.5 was easily purified by flash chromatography to deliver pure decarboxylation product in

45% yield.

O O N S S Cl C C Cl ONa O RCOOH RCOCl DMAP N O C R

S SnBu3 HSnBu3 N + CO2 + RH AIBN, heat Scheme 7.2. General scheme of Barton decarboxylation.

7.2.4. Condensation of an alkylhydrazine with a β-ketoaldehyde. An alternative was

explored to generate the requisite N-substituted pyrazole by condensation of an

alkylhydrazine with a β-ketoaldehyde (Scheme 7.3). However, a mixture of 3- and

227 5-substituted isomers usually occurs. The failure to isolate the individual isomers led

early workers to the conclusion that these two forms are indistinguishable and that the 3-

and 5-positions are equivalent.37 Actually, the isomers are separatable using normal flash chromatography. The problem with this route consists of two factors: (1) 2-heptanone condenses with ethyl formate at both the methyl and the methylene positions to provide a mixture of sodium salts of hydroxymethylene ketones (7.7 and the isomer);30, 38 and (2)

7.7 readily undergoes self-condensation to form a symmetrical 1,3,5-triacylbenzene.39-41

As a consequence of the above factors, the yield of 7.5 through this route was low (10%).

O O OH O OH 1. Na, ether. 2. HCl + HCOOC H + C H CH 2 5 C5H11 C H H C C H 5 11 3 crude: 45% H 3 C H 7.7 4 9 C5H11 O OH NH2-NH-(CH2)4CH=CH2 N + N C5H11 N N C5H11 C H ethanol H (CH2)4CH=CH2 (CH2)4CH=CH2 7.5 (10%) 7.10 (11%) ether + 1/3 PBr Br-(CH ) CH=CH HO-(CH2)4CH=CH2 3 -78 °C, 50% 2 4 2 7.8 MeOH, 50 hr NH2-NH2 · H2O + Br-(CH2)4CH=CH2 NH2-NH-(CH2)4CH=CH2 60% 7.9 Scheme 7.3. An alternative route for synthesis of pyrazole 7.5.

7.2.5. Attempted ozonolysis off an alkenylpyrazole. Oxidation of an alkene to an

aldehyde is generally accomplished by ozonolysis. However, the ozonolysis of pyrazole

7.5 encountered problems. Apparently, the pyrazole ring is also oxidized. After trying the ozonolysis three times, I failed to get the expected pyrazole aldehyde 7.6 (Scheme 7.4).

Since the double bond on the side chain is supposed to be more readily oxidized than the

228 double bonds on the aromatic ring, it may be possible to control consumption of ozone by

careful TLC monitoring of the reaction.

7.2.6. A strategy for conjugation of the isostere hapten with peptides. Sodium

cyanoborohydride is generally useful for imine reduction and reductive amination of

proteins in aqueous solution at pH 6 - 8.42 It is anticipated that the aldehyde 7.6 can be coupled with the dipeptide N-acetyl- gly-lys-OMe or bovine serum albumin (BSA) by reductive alkylation in methanol (Scheme 7.4).

N (1),ozone, MeOH N NaBH CN, HOAc N N 3 C5H11 C5H11 N C5H11 N (CH ) CH=CH -78 °C, (2), Me S (CH ) CHO protein protein 2 4 2 2 2 4 NH2 (CH2)5NH 7.5 7.6 protein=Actyl-Gly-Lys-OMe or BSA

Scheme 7.4. Reductive amination of proteins with pyrazole aldehyde 7.6 that is to be generated by ozonolysis of alkene 7.5.

229 7.3. Experimental Procedures.

Preparation of 2-hydroxy-4-oxo-non-2-enoic acid ethyl ester (7.1). To a suspension of

metallic sodium (1.06 g, 46.08 mmol ) in freshly distilled ethanol (50 mL) was slowly

added dropwise a mixture of 2-heptanone (4.18 g, 36.6 mmol) and diethyl oxalate (6.4 g,

43.8 mmol). After being stirred at 60 °C for 9 h, the mixture was treated with 3 N HCl to

adjust the pH to 5. The cloudy mixture was extracted with ethyl acetate (3 x 15 mL). The

red-brown organic layer was washed with brine, dried with sodium sulphate and

concentrated to obtain the crude product. The crude compound was purified by silica gel

chromatography (15 % ethyl acetate in hexane, TLC: Rf = 0.3) to deliver 5.02 g pure 7.1

1 as a red-brown oil. H NMR (CDCl3, 300 MHz) δ 6.36 (s, 1H), 4.36 (q, J = 7.14 Hz, 2H),

2.48 (t, J = 7.4 Hz, 2H), 1.65 (m, 2H), 1.37 (t, J = 7.14 Hz, 3H), 1.34-1.29 (4H), 0.90 (t, J

13 = 6.87 Hz, 3H). C NMR (CDCl3, 50 MHz, APT) δ 203.42 (+) (CO), 166.69 (+) (COH),

162.22 (+) (COO), 101.66 (-) (CH), 62.53 (+) (CH2), 40.94 (+) (CH2), 31.33 (+) (CH2),

24.62 (+) (CH2), 22.42 (+) (CH2), 14.10 (-) (CH3), 13.93 (-) (CH3).

5-Pentyl-1H-pyrazole-3-carboxylic acid ethyl ester (7.2). Hydrazine hydrate (1.65 mL,

0.033 mol) was slowly added to a solution of 7.1 (4.61 g, 0.0215 mol) in ethanol (10 mL)

with acetic acid (4 mL) under ice-cooling. The mixture was then heated for 24 h under

reflux, then cooled, neutralized with sat. NaHCO3 solution, and extracted with ethyl

acetate. The ethyl acetate extract was washed with saturated NaHCO3 solution, dried with

Na2SO4, and evaporated to dryness. The crude residue was purified by silica gel chromatography (25 % ethyl acetate in hexane, TLC: Rf = 0.3) to give 3.8 g (84 %) of

230 1 pure 7.2. H NMR (CDCl3, 300 MHz) δ 6.60 (s, 1H), 4.36 (q, J = 7.1 Hz, 2H), 2.68 (t, J =

11.2 Hz, 2H), 1.65 (m, 2H), 1.36 (t, J = 7.1 Hz, 3H), 1.32-1.27 (4H), 0.90 (t, J = 10.6 Hz,

13 3H). C NMR (CDCl3, 50 MHz) δ 162.25 (COO), 146.73 (C), 142.07 (C), 105.68 (CH),

60.39 (CH2), 31.04 (CH2), 28.52 (CH2), 25.38 (CH2), 22.08 (CH2), 13.94 (CH3), 13.66

+ (CH3). HRMS (EI) (m/z) calcd for C11H18N2O2 (M ) 210.1368, found 210.1371; calcd for

+ C10H15N2O2 (M -CH3) 195.1134, found 195.1132.

Preparation of 6-iodohexene (7.3). 6-Chlorohexene (500 mg, 4.22 mmol) and sodium

iodide (3.8 g, 25.3 mmol) were dissolved in acetone (28 mL). The solution was stirred for

9 h under reflux. Insoluble sodium chloride was generated as the reaction proceeded. The

mixture was then cooled, evaporated to remove acetone. Water (10 mL) was added to

dissolve the salt. The mixture was extracted with ethyl acetate (3 x 15 mL). The brown

extract was washed with Na2S2O3 and brine, dried with Na2SO4, and evaporated to give

1 650 mg (3.1 mmol, 73 %) 7.3 without further purification. H NMR (CDCl3, 300 MHz) δ

5.77 (m, 1H), 5.0 (m, 2H), 3.2 (t, J = 5.1 Hz, 2H), 2.08 (m, 2H), 1.84 (m, 2H), 1.52 (m,

2H).

Preparation of 1-hex-5-enyl-5-pentyl-1H-pyrazole-3-carboxylic acid (7.4). Pyrazole

7.2 (565 mg, 2.69 mmol) in toluene (5 mL) was added dropwise to a stirred suspension of

NaH (60 % in oil) (143 mg, 3.58 mmol) in toluene (10 mL) at room temperature. The resulting mixture was stirred at 60 °C for 1 h, then 6-iodohexene (650 mg, 3.1 mmol) was added and the resulting mixture was heated at 100 °C overnight. Solvent was removed by evaporation, and water was added to the residue. The mixture was extracted with EtOAc

231 (3 x 10 mL). The organic layer was washed with brine, dried with Na2SO4, and evaporated to give crude ethyl ester (593 mg). The ester (593 mg) was then dissolved in a solution of NaOH (1.5 N, 6 mL) in ethanol (10 mL), and refluxed for 3 h. After removal of the solvent, the residue was acidified with 3 N HCl to pH = 4.0, and then extracted with EtOAc (3 x 10 mL). The organic layer was washed with brine, dried with Na2SO4, and concentrated. The crude acid was purified by silica gel chromatography (1 % acetic

acid in ethyl acetate, TLC: Rf = 0.23) to give 353 mg (50 %) of pure 7.4 as light yellow

1 crystals (melting point: 145°C). H NMR (CDCl3, 300 MHz) δ 6.63 (s, 1H), 5.77 (m, 1H),

4.99 (m, 2H), 4.11 (t, J = 11.1 Hz, 2H), 2.59 (t, J = 11.0 Hz, 2H), 2.10 (m, 2H), 1.86 (m,

13 2H), 1.67 (m, 2H), 1.45-1.3 (6H), 0.92 (t, J = 10.4 Hz, 3H). C NMR (CDCl3, 50 MHz,

APT) δ 166.00 (+) (COO), 144.68 (+) (C), 141.69 (+) (C), 137.98 (-) (CH), 114.89 (+)

(CH2), 107.37 (-) (CH), 49.52 (+) (CH2), 33.15 (+) (CH2), 31.25 (+) (CH2), 29.65 (+)

(CH2), 27.93 (+) (CH2), 25.70 (+) (CH2), 25.28 (+) (CH2), 22.33 (+) (CH2), 13.85 (-)

(CH3).

Preparation of 1-hex-5-enyl-5-pentyl-1H-pyrazole (7.5). Acid 7.4 (140 mg, 0.53 mmol)

was put into an evacuated flask and heated under 170 °C for 9 h using a heating mantle

filled with sand. The dark residue was purified by silica gel chromatography (10 % ethyl

1 acetate in hexane, TLC: Rf = 0.2) to afford (53 mg, 45%) of pure 7.5. H NMR (CDCl3,

400 MHz) δ 7.38 (d, J = 1.6 Hz, 1H), 5.98 (d, J = 1.6 Hz, 1H), 5.76 (m, 1H), 5.01-4.93

(m, 2H), 4.01 (t, J = 7.2 Hz, 2H), 2.56 (t, J = 7.6 Hz, 2H), 2.07 (m, 2H), 1.82 (m, 2H),

13 1.63 (m, 2H), 1.44-1.34 (6H), 0.92 (t, J = 7.2 Hz, 3H). C NMR (CDCl3, 100 MHz) δ

232 142.52 (C), 138.21 (CH), 138.05 (CH), 114.81 (C), 103.65 (CH), 48.62 (CH2), 33.24

(CH2), 31.44 (CH2), 29.73 (CH2), 28.29 (CH2), 25.90 (CH2), 25.36 (CH2), 22.37 (CH2),

+ 13.92 (CH3). HRMS (FAB) (m/z) calcd for C14H25N2 (MH ) 221.2018, found 221.2027.

Ozonolysis. Alkene 7.5 (17 mg, 0.078 mmol) was dissolved in methanol (10 mL) and the

solution was cooled to -78 °C. Ozone, generated from a cylinder of oxygen with a

Welsbach ozonizer operated with an O2 pressure of 6 lb per square inch and a voltage

setting of 58 v, was bubbled through the methanol solution at -78 °C until a blue color

appeared and persisted for 5 minutes. The excess of ozone was then flushed away with

nitrogen. While still at -78 °C, Me2S (1 mL) was added and the solution was allowed to warm gradually to room temperature. The solvent was removed by rotary evaporation. A

1H NMR spectrum of the mixture showed the disappearance of protons on the pyrazole

double bonds. Thus, the desired aldehyde pyrazole was not obtained.

Preparation of 1-hydroxyoct-1-en-3-one (7.7). To a suspension of metallic sodium (201

mg, 8.75 mmol ) in freshly distilled ether (25 mL) was slowly added dropwise a mixture

of 2-heptanone (1 g, 8.75 mmol) and ethyl formate (648 mg, 8.75 mmol) with ice bath

cooling. The mixture became a slurry after being stirred for 9 h at room temperature. The slurry was poured into cold water (10 mL). The mixture was separated. The aqueous

layer was washed with ether twice and then acidified with 3 N HCl to pH = 5. The

resulting mixture was extracted with ethyl acetate (3 x 10 mL). The light yellow organic

layer was washed with brine, dried with sodium sulphate and concentrated to obtain 561

mg of the crude product 7.7 (45 %) which was used without further purification. 1H NMR

233 (CDCl3, 300 MHz) δ 7.9 (d, J = 4.8 Hz, 1H), 5.52 (d, J = 4.8 Hz, 1H), 2.35 (t, J = 7.4 Hz,

2H), 1.6 (m, 2H), 1.4-1.2 (4H), 0.90 (t, J = 6.87 Hz, 3H).

Preparation of 6-bromohexene (7.8).43 To a solution of 5-hexen-1-ol (3.41 g, 34.1 mmol) in freshly distilled ether (25 mL) under argon at -78 °C was added dropwise a solution of phosphorus tribromide (3.54 g, 13.7 mmol) in ether (5 mL). The reaction mixture was slowly allowed to warm to room temperature overnight, whereupon water (5 mL) and ether (15 mL) were added. The mixture was washed with an aqueous solution of saturated potassium carbonate (10 mL). After drying and removal of the solvents, the residue was distilled in vacuo to afford 2.78 g (50 %) of the hexenyl bromide, bp 34-36

1 °C (0.05 mm). H NMR (CDCl3, 300 MHz) δ 5.77 (m, 1H), 5.0 (m, 2H), 3.4 (t, J = 7.4

Hz, 2H), 2.1 (m, 2H), 1.88 (m, 2H), 1.55 (m, 2H).

Preparation of 5-hexen-1-yl-hydrazine (7.9). Hydrazine hydrate (10 mL, 0.2 mol) was added to 6-bromohexene (2.78 g, 17.05 mmol) in methanol (10 mL). The cloudy system

was flushed with argon and stirred for 50 h at room temperature. The solution was then

extracted three times with ether (10 mL). The combined extracts were dried over Na2SO4.

After removal of solvent by evaporation, the crude residue was purified by silica gel chromatography (20 % ethyl acetate in hexane, TLC: Rf = 0.2) to afford (1.1 g, 60%) of

1 pure 7.9. H NMR (CDCl3, 300 MHz) δ 5.77 (m, 1H), 5.0 (m, 2H), 2.9 (b, s, 3H), 2.77 (t,

J = 7.4 Hz, 2H), 2.05 (m, 2H), 1.6-1.4 (4H).

Preparation of 1-hex-5-enyl-5-pentyl-1H-pyrazole (7.5) and 1-hex-5-enyl-3-pentyl

-1H-pyrazole (7.10). Hydrazine 7.9 (495 mg, 4.35 mmol) was slowly added to a solution

234 of 7.7 (561 mg, 3.95 mmol) in ethanol (10 mL) and acetic acid (4 mL) under ice-cooling.

The mixture was heated for 24 h under reflux, then cooled, neutralized with saturated

NaHCO3 solution, and extracted with ethyl acetate. The ethyl acetate extract was washed with saturated NaHCO3 solution, dried with Na2SO4, and evaporated to dryness. The

crude residue was purified by silica gel chromatography (10 % ethyl acetate in hexane) to

1 give pure 7.5 (84 mg, Rf = 0.3, 10%) and pure 7.10 (98 mg, Rf = 0.35, 11 %). H NMR for

1 7.5 is displayed above. H NMR for 7.10 (CDCl3, 400 MHz) δ 7.18 (d, J = 2.4 Hz, 1H),

5.93 (d, J = 2.4 Hz, 1H), 5.71 (m, 1H), 4.95-4.85 (m, 2H), 3.98 (t, J = 7.2 Hz, 2H), 2.54 (t,

J = 7.6 Hz, 2H), 2.00 (m, 2H), 1.78 (m, 2H), 1.56 (m, 2H), 1.35-1.24 (6H), 0.82 (t, J =

7.2 Hz, 3H).

235 7.4. References

1. Amarnath, V.; Amarnath, K.; Valentine, W. M.; Eng, M. A.; Graham, D. G.,

Intermediates in the Paal-Knorr synthesis of pyrroles. 4-Oxoaldehydes. Chem. Res.

Toxicol. 1995, 8, (2), 234-8.

2. Bleasdale, C.; Golding, B. T.; Kennedy, G.; MacGregor, J. O.; Watson, W. P.,

Reactions of muconaldehyde isomers with nucleophiles including tri-O-acetylguanosine: formation of 1,2-disubstituted pyrroles from reactions of the (Z,Z)-isomer with primary amines. Chem. Res. Toxicol. 1993, 6, (4), 407-12.

3. DeCaprio, A. P.; Olajos, E. J.; Weber, P., Covalent binding of a neurotoxic n-hexane metabolite: conversion of primary amines to substituted pyrrole adducts by

2,5-hexanedione. Toxicol Appl. Pharmacol. 1982, 65, (3), 440-50.

4. Graham, D. G.; Anthony, D. C.; Boekelheide, K.; Maschmann, N. A.; Richards, R.

G.; Wolfram, J. W.; Shaw, B. R., Studies of the molecular pathogenesis of hexane neuropathy. II. Evidence that pyrrole derivatization of lysyl residues leads to protein crosslinking. Toxicol. Appl. Pharmacol. 1982, 64, (3), 415-22.

5. Schauenstein, E.; Esterbauer, H., Formation and properties of reactive aldehydes.

Ciba Foundation symposium 1978, (67), 225-44.

6. Benedetti, A.; Comporti, M.; Esterbauer, H., Identification of 4-hydroxynonenal as a cytotoxic product originating from the peroxidation of liver microsomal lipids. Biochim.

Biophy. Acta. 1980, 620, (2), 281-96.

236 7. Sayre, L. M.; Sha, W.; Xu, G.; Kaur, K.; Nadkarni, D.; Subbanagounder, G.;

Salomon, R. G., Immunochemical evidence supporting 2-pentylpyrrole formation on

proteins exposed to 4-hydroxy-2-nonenal. Chem. Res. Toxicol. 1996, 9, (7), 1194-201.

8. Witz, G., Biological interactions of alpha,beta-unsaturated aldehydes. Free Radic.

Biol. Med. 1989, 7, (3), 333-49.

9. Esterbauer, H.; Schaur, R. J.; Zollner, H., Chemistry and biochemistry of

4-hydroxynonenal, malonaldehyde and related aldehydes. Free Radic. Biol. Med. 1991,

11, (1), 81-128.

10. Sayre, L. M.; Arora, P. K.; Iyer, R. S.; Salomon, R. G., Pyrrole formation from

4-hydroxynonenal and primary amines. Chem. Res. Toxicol. 1993, 6, (1), 19-22.

11. Iyer, R. S.; Ghosh, S.; Salomon, R. G., Levuglandin E2 crosslinks proteins.

Prostaglandins 1989, 37, (4), 471-80.

12. DiFranco, E.; Subbanagounder, G.; Kim, S.; Murthi, K.; Taneda, S.; Monnier, V. M.;

Salomon, R. G., Formation and stability of pyrrole adducts in the reaction of levuglandin

E2 with proteins. Chem. Res. Toxicol. 1995, 8, (1), 61-7.

13. Salomon, R. G.; Miller, D. B.; Zagorski, M. G.; and Coughlin, D. J., Solvent induced

fragmentation of prostaglandin endoperoxides. New aldehyde products from PGH2 and a

novel intramolecular 1,2-hydride shift during endoperoxide fragmentation in aqueous solution. J. Am. Chem. Soc. 1984, 106, 6049-6060.

14. Brame, C. J.; Salomon, R. G.; Morrow, J. D.; Roberts, L. J., 2nd, Identification of extremely reactive gamma-ketoaldehydes (isolevuglandins) as products of the isoprostane

237 pathway and characterization of their lysyl protein adducts. J. Biol. Chem. 1999, 274,

(19), 13139-46.

15. Roberts, L. J., 2nd; Brame, C. J.; Chen, Y.; Morrow, J. D.; Salomon, R. G.,

Formation of reactive products of the isoprostane pathway: isolevuglandins and cyclopentenone isoprostanes. Adv. Exp. Med. Biol. 1999, 469, 335-41.

16. Roberts, L. J., 2nd; Salomon, R. G.; Morrow, J. D.; Brame, C. J., New developments in the isoprostane pathway: identification of novel highly reactive gamma-ketoaldehydes

(isolevuglandins) and characterization of their protein adducts. Faseb J. 1999, 13, (10),

1157-68.

17. Salomon, R. G.; Kaur, K.; Podrez, E.; Hoff, H. F.; Krushinsky, A. V.; Sayre, L. M.,

HNE-derived 2-pentylpyrroles are generated during oxidation of LDL, are more

prevalent in blood plasma from patients with renal disease or atherosclerosis, and are

present in atherosclerotic plaques. Chem. Res. Toxicol. 2000, 13, (7), 557-64.

18. Salomon, R. G.; Sha, W.; Brame, C.; Kaur, K.; Subbanagounder, G.; O'Neil, J.; Hoff,

H. F.; Roberts, L. J., 2nd, Protein adducts of iso[4]levuglandin E2, a product of the isoprostane pathway, in oxidized low density lipoprotein. J. Biol. Chem. 1999, 274, (29),

20271-80.

19. Gossauer, A.; Nesvadba, P., Oxidation and reduction of the pyrrole ring. . Pyrroles

Part One: The Synthesis and the Physical Aspects of the Pyrrole Ring (Jones, R. A., Ed.)

Wiley, New York. 1990, 499-536.

238 20. Gardini, G. P., The oxidation of monocyclic pyrroles. Adv. Heterocycl. Chem. 1973,

15, 67-99.

21. Smith, E. B.; Jensen, H. B., Autoxidation of three 1-alkylpyrroles. J. Org. Chem.

1967, 32, 3330-3334.

22. Hoft, E.; Katritzky, A. R.; Nesbit, M. R., The autoxidation of alkylpyrroles.

Tetrahedron Lett. 1967, 32, 3041-3044.

23. Katritzky, A. R.; Nesbit, M. R., The autoxidation of alkylpyrroles. Tetrahedron Lett.

1967, 32, 3041-3044.

24. DeCaprio, A. P., Mechanisms of in vitro pyrrole adduct autoxidation in

2,5-hexanedione-treated protein. Mol. Pharmacol. 1986, 30, (5), 452-8.

25. Zhu, M.; Spink, D. C.; Yan, B.; Bank, S.; DeCaprio, A. P., Formation and structure of cross-linking and monomeric pyrrole autoxidation products in 2,5-hexanedione-treated amino acids, peptides, and protein. Chem. Res. Toxicol. 1994, 7, (4), 551-8.

26. Anthony, D. C.; Amarnath, V.; Graham, D. G., Pyrrole-mediated protein cross-linking: Toward an understanding of the molecular mechanism. Toxicologist 1990,

10, 183.

27. Kobierski, M. E.; Kim, S.; Murthi, K. K.; Iyer, R. S.; Salomon, R. G., Synthesis of a pyrazole isostere of pyrroles formed by the reaction of the ε-amino groups of protein lysyl residues with levuglandin E2. J. Org. Chem. 1994, 59, 6044-6050.

239 28. Harris, T. M.; Boatman, S.; Hauser, C. R., Alkylations at the γ−position of acetoacetaldehyde and α−benzylacetoacetaldehyde through their dicarbanions. J. Am.

Chem. Soc. 1963, 85, 3273-3276.

29. Mariella, R. P., Condensations of unsymmetrical ketones. Ι. Condensations with ethyl formate. J. Am. Chem. Soc. 1947, 69, 2670-2672.

30. Earl Royals, E. C., E. R. , Hydroxymethylene ketones. IV. Orientation in the condensation of methyl n-hexyl ketone with methyl formate. J. Am. Chem. Soc. 1955, 77,

3155-3157.

31. Bernard, A.; Cocco, M. T.; Maccioni, A.; Plumitallo, A., Phytotoxic activity in pyrazole derivatives. Farmaco. 1985, 40, 259-271.

32. Elguero, J., Comprehensive Heterocyclic Chemistry. Pergamon Press: Oxford. 1996,

3, 1-75.

33. Kano, K.; Scarpetti, D.; Warner, J. C.; Anselme, J. P.; Springer, J. P.; Arison, B. H.,

Benzoylphenyl-1-methylpyrazoles. Can. J. Chem. 1986, 64, 2211-2219.

34. Jones, G. B.; Chapman, B. J., Decarboxylation of indole-2-carboxylic acids:

Improved procedures. . J. Org. Chem. 1993, 58, 5558-5559.

35. Barton, D. H. R.; Crich, D.; Motherwell, W. B., The invention of new radical chain reactions. Part VIII. Radical chemistry of thiohydroxamic esters; A new method for the generation of carbon radicals from carboxylic acids. Tetrahedron 1985, 41, 3901-3924.

36. Barton, D. H. R., The invention of chemical reactions of relevance to the chemistry of natural products. Pure & Appl. Chem. 1994, 66, 1943-1954.

240 37. Habraken, C. L.; Moore, J. A., Heterocyclic studies. XVI. The assignment of isomeric and tautomeric structures of pyrazoles by nuclear magnetic resonance. J. Org.

Chem. 1965, 30, 1892-1896.

38. Cason, J.; Chang, M. P., Condensation of acrylonitrile with unsymmetrical ketones. J.

Org. Chem. 1956, 21, 449-454.

39. Eapen, K. C.; Tamborski, C., Synthesis of 1,3,5-tri-n-alkylbenzene compounds. J.

Org. Chem. 1988, 53, 5564-5567.

40. Kaushal, R.; Sorani, S.; Deshpande, S. S., J. Ind. Chem. Soc. 1942, 19, 107.

41. Mariella, R. P.; Godar, E., Notes - Condensations of unsymmetrical ketones. IV.

Participation of methyl and methylene groups in condensation reactions. J. Org. Chem.

1957, 22, 566-568.

42. Hayase, F.; Nagaraj, R. H.; Miyata, S.; MNjoroge, F. G.; Monnier, V. M., J. Biol.

Chem. 1989, 264, 3758.

43. Samsel, E. G.; Kochi, J. K., Oxidative alkylation of cobalt complexes with hydrazines. Inorg. Chem. 1986, 25, 2450-2457.

241

Appendix

242 O O OMe

O O

1 Figure S1. H NMR (200 MHz, CDCl3) spectrum of methyl 6-(2,5-dioxolanyl)-4 -oxohexanoate (2.1).

O O OMe

O O

13 Figure S2. C NMR (50 MHz, CDCl3) spectrum of methyl 6-(2,5-dioxolanyl)- -4-oxohexanoate (2.1).

243 O O OH

O O

1 Figure S3. H NMR (200 MHz, CDCl3) spectrum of 6-(2,5-dioxolanyl)-4 -oxohexanoic acid (2.2).

O O OH

O O

13 Figure S4. C NMR (100 MHz, CDCl3) spectrum of 6-(2,5-dioxolanyl)-4- oxohexanoic acid (2.2).

244

O O O

O O

1 Figure S5. H NMR (200 MHz, CDCl3) spectrum of (2.3).

O O O O O

13 Figure S6. C NMR (50 MHz, CDCl3) spectrum of (2.3). C, CH2 (-); CH, CH3 (+).

245 O O O

O

1 Figure S7. H NMR (200 MHz, CDCl3) spectrum of DOHAFm (2.4).

O O O

O

13 Figure S8. C NMR (100 MHz, CDCl3) spectrum of DOHAFm (2.4).

246

H CO O 3 O H N N N H O O

O

1 Figure S9. H NMR (400 MHz, CDCl3) spectrum of (2.5).

H3CO O O H N N N H O O

O

13 Figure S10. C NMR (100 MHz, CDCl3) spectrum of (2.5).

247 O H H3CO O N N O N H

O O H

1 Figure S11. H NMR (400 MHz, CDCl3) spectrum of (2.6).

O H H3CO O N

N O N H

O O H

13 Figure S12. C NMR (150 MHz, CDCl3) spectrum of (2.6).

248 O N O 3

O OH

1 Figure S13. H NMR (400 MHz, CDCl3) spectrum of (2.9).

O N O 3

O OH

13 Figure S14. C NMR (100 MHz, CDCl3) spectrum of (2.9).

249 N O O

3 O O O N O

1 Figure S15. H NMR (400 MHz, CDCl3) spectrum of CEPFmSu (2.10).

N O O

3 O O O N O

13 Figure S16. C NMR (100 MHz, CDCl3) spectrum of CEPFmSu (2.10).

250

O

NH NH O H N O C N C S H O

1 Figure S17. H NMR (400 MHz, CD3OD) spectrum of (2.18).

O

NH NH O NH2 N S H

1 Figure S18. H NMR (200 MHz, CD3OD) spectrum of (2.19).

251 O

NH NH O N O

N O S H

1 Figure S19. H NMR (200 MHz, CDCl3) spectrum of (2.20).

O

NH NH O N N S H O

OH

1 Figure S20. H NMR (400 MHz, CD3OD) spectrum of (2.21).

252 O O OMe

O

1 Figure S21. H NMR (200 MHz, CDCl3) spectrum of (2.22).

O

NH NH O

N 4 NH2 S H

1 Figure S22. H NMR (400 MHz, CD3OD) spectrum of (2.23).

253 O

NH NH O

N 4 NH2 S H

13 Figure S23. C NMR (100 MHz, CD3OD) spectrum of (2.23).

O

NH NH O N N 4 S H O

OMe

1 Figure S24. H NMR (400 MHz, CD3OD) spectrum of (2.24).

254

O

NH NH O N N 4 S H O

OH

1 Figure S25. H NMR (400 MHz, CD3OD) spectrum of (2.25).

O

NH NH O N N 4 S H O

OH

13 Figure S26. C NMR (100 MHz, CD3OD) spectrum of (2.25).

255 O O CO O P O O 7 O C8H17 O N O C14H29 O

1 Figure S27. H NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.26).

O O CO O P O O 7 O C8H17 O N O C14H29 O

13 Figure S28. C NMR (50 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.26).

256 O O CO O P O OH 7 O C8H17 O N O C14H29 O

1 Figure S29. H NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.27).

O O CO O P O OH 7 O C8H17 O N O C14H29 O

13 Figure S30. C NMR (100 MHz, CDCl3:CD3OD = 1:1) spectrum of (2.27).

257 COO

O O P N HO O OH

O C14H29 O

1 Figure S31. H NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.28).

COO

O O P N HO O OH

O C14H29 O

13 Figure S32. C NMR (400 MHz, CDCl3 : CD3OD = 1 : 1) spectrum of (2.28).

258 COOH

O O P N HO O OH

O C14H29 O

1 Figure S33. H NMR (400 MHz, CDCl3) spectrum of (2.29).

COOH

O O P N HO O OH

O C14H29 O

13 Figure S34. C NMR (100 MHz, CDCl3 :CD3OD:D2O = 50:50:1) spectrum of (2.29).

259

HO H O N O O O O N

1 Figure S35. H NMR (400 MHz, CDCl3) spectrum of (2.30).

HO H O N O O O O N

13 Figure S36. C NMR (100 MHz, CDCl3) spectrum of (2.30).

260

F F F F O F H O N O O O O N

1 Figure S37. H NMR (400 MHz, CDCl3) spectrum of (2.31).

F F

F F

O F H O N O O O O N

13 Figure S38. C NMR (100 MHz, CDCl3) spectrum of (2.31).

261 A

B

C

262 D

E

Figure S39. Tandem MS characterization of CEP modified HSA shows series of

fragment ions sufficient to identify CEP modifications on lysyl residues of the HSA peptides. (A). MS/MS spectrum of the doubly charged ion m/z 589.266. (B). MS/MS spectrum of the doubly charged ion m/z 674.762. (C). MS/MS spectrum of the doubly charged ion m/z 881.401. (D). MS/MS spectrum of the doubly charged ion m/z 687.784.

(E). MS/MS spectrum of the doubly charged ion m/z 636.252. Asterisks denote fragment ions with the modified lysyl residue.

263 A

B

C

264 D

E

F

265 G

H

Figure S40. Tandem MS characterization of CEP modified MSA. (A). MS/MS spectrum of the doubly charged ion m/z 650.3296. (B). MS/MS spectrum of the doubly charged ion m/z 1047.0802. (C). MS/MS spectrum of the doubly charged ion m/z 769.4069. (D).

MS/MS spectrum of the doubly charged ion m/z 612.8407. (E). MS/MS spectrum of the doubly charged ion m/z 872.9611. (F). MS/MS spectrum of the triplet charged ion m/z

849.4302. (G). MS/MS spectrum of the triplet charged ion m/z 857.4633. (H). MS/MS spectrum of the triplet charged ion m/z 694.6888. Asterisks denote fragment ions with the modified lysyl residue.

266 A

B

C

267 D

E

F

268 G

Figure S41. Tandem MS characterization of CEP modified CEO. (A). MS/MS spectrum

of the doubly charged ion m/z 540.2886. (B). MS/MS spectrum of the triplet charged ion

m/z 801.7021. (C). MS/MS spectrum of the doubly charged ion m/z 839.3332. (D).

MS/MS spectrum of the triplet charged ion m/z 808.0259. (E). MS/MS spectrum of the doubly charged ion m/z 610.2589. (F). MS/MS spectrum of the doubly charged ion m/z

630.8334. (G). MS/MS spectrum of the doubly charged ion m/z 459.1887. Asterisks

denote fragment ions with the modified lysyl residue.

269 A

B

C

270 D

E

271 F

Figure S42. Tandem MS characterization of CEP modified myoglobin. (A). MS/MS spectrum of the doubly charged ion m/z 671.2667. (B). MS/MS spectrum of the doubly charged ion m/z 604.9635. (C). MS/MS spectrum of the doubly charged ion m/z

815.2087. (D). MS/MS spectrum of the quartet charged ion m/z 557.4288. (E). MS/MS spectrum of the doubly charged ion m/z 785.6061. (F). MS/MS spectrum of the doubly charged ion m/z 899.6666. Asterisks denote fragment ions with the modified lysyl residue.

272 A

B

C

273 D

E

Figure S43. Tandem MS characterization of CEP modified GPDH. (A). MS/MS spectrum of the doubly charged ion m/z 655.3343. (B). MS/MS spectrum of the doubly charged ion m/z 669.3289. (C). MS/MS spectrum of the doubly charged ion m/z

923.9847. (D). MS/MS spectrum of the doubly charged ion m/z 639.3413. (E). MS/MS spectrum of the doubly charged ion m/z 740.8362. Asterisks denote fragment ions with the modified lysyl residue.

274 A

B

275 C

D

276 E

F

G

277 H

Figure S44. Tandem MS characterization of CEPH modified BSA. (A). MS/MS spectrum of the triplet charged ion m/z 594.9934. (B). MS/MS spectrum of the doubly charged ion m/z 541.8060. (C). MS/MS spectrum of the doubly charged ion m/z

937.9577. (D). MS/MS spectrum of the doubly charged ion m/z 526.7835. (E). MS/MS spectrum of the doubly charged ion m/z 612.3604. (F). MS/MS spectrum of the doubly charged ion m/z 689.4319. (G). MS/MS spectrum of the doubly charged ion m/z

765.3984. (H). MS/MS spectrum of the doubly charged ion m/z 964.5088. Asterisks denote fragment ions with the modified lysyl residue.

278 A

B

C

279 D

Figure S45. Tandem MS characterization of CEPH modified BSA. (A). MS/MS spectrum of the doubly charged ion m/z 512.2829. (B). MS/MS spectrum of the doubly charged ion m/z 627.8604. (C). MS/MS spectrum of the doubly charged ion m/z

608.8177. (D). MS/MS spectrum of the doubly charged ion m/z 732.3707. Asterisks denote fragment ions with the modified lysyl residue.

280

Figure S46. Tandem MS characterization of the doubly charged ion m/z 596.8715 from a

MS scan of tryptic digested CEPH modified CEO. Asterisks denote fragment ions with the modified lysyl residue.

281 A

B

C

282 D

E

F

283 G

H

I

284 J

K

L

285 M

Figure S47. Tandem MS characterization of CEPH modified GPDH. (A). MS/MS spectrum of the doubly charged ion m/z 635.0367. (B). MS/MS spectrum of the doubly charged ion m/z 712.5500. (C). MS/MS spectrum of the doubly charged ion m/z

919.7650. (D). MS/MS spectrum of the doubly charged ion m/z 572.9763. (E). MS/MS spectrum of the triplet charged ion m/z 899.6923. (F). MS/MS spectrum of the doubly charged ion m/z 726.0422. (G). MS/MS spectrum of the doubly charged ion m/z

980.7797. (H). MS/MS spectrum of the doubly charged ion m/z 731.1005. (I). MS/MS spectrum of the doubly charged ion m/z 941.7422. (J). MS/MS spectrum of the triplet charged ion m/z 579.9560. (K). MS/MS spectrum of the doubly charged ion m/z

508.9000. (L). MS/MS spectrum of the doubly charged ion m/z 696.1018. (M). MS/MS spectrum of the doubly charged ion m/z 797.5823. Asterisks denote fragment ions with the modified lysyl residue.

286 A

9 8 7 6 5 4 3 1 y

D A H K S E V A H R

b 3 4 6 7

B

14 11 10 9 8 y

K V P Q V S T P T L V E V S R

b 2 5

287 C 8 7 6 5 4 y

A F K A W A V A R b 3

10 9 8 7 y D F P K A E F A E V S K

b 3 5 8

288 E 10 9 8 7 y F K D L G E E N F K

b 3

Figure S48. Tandem MS characterization of CEPFmHSA revealed five CEP modifications. (A). MS/MS spectrum of the doubly charged ion m/z 636.248. (B).

MS/MS spectrum of the doubly charged ion m/z 881.408. (C). MS/MS spectrum of the doubly charged ion m/z 571.262. (D). MS/MS spectrum of the doubly charged ion m/z

687.787. (E). MS/MS spectrum of the doubly charged ion m/z 674.765.

289

A

9 8 7 6 5 4 1 y

D A H K S E V A H R

b 3 4 5 6 7

B

8 6 5 4 3 2 y

A F K A W A V A R

b 4

290

C 10 9 8 7 6 5 3 y

F P K A E F A E V S K

b 2

Figure S49. Tandem MS characterization of CEPFmHSA revealed five CEPFm modifications. (A). MS/MS spectrum of the doubly charged ion m/z 725.281. (B).

MS/MS spectrum of the doubly charged ion m/z 660.289. (C). MS/MS spectrum of the doubly charged ion m/z 776.820.

291 Table S1. Total iron levels of patients treated with vitamin Eα or placebo at baseline, three months, six months and nine months. Patient code VE treatment/ Baseline 3 months 6 months 9 months placebo P1 placebo 67 54 57 53 T1 treatment 57 48 41 60 T2 treatment 43 60 34 41 P2 placebo 50 42 62 75 P3 placebo 43 39 65 P4 placebo 42 39 41 61 T3 treatment 48 54 69 98 T4 treatment 60 46 62 P5 placebo 62 112 74 69 T5 treatment 49 48 53 25 T6 treatment 37 64 71 27 P6 placebo 52 58 62 27 P7 placebo 69 64 74 93 P8 placebo 70 64 57 56 T7 treatment 72 107 98 T8 treatment 58 89 56 46 T9 treatment 47 57 42 65 P9 placebo 41 46 45 42 P10 placebo 69 110 55 91 T10 treatment 49 32 38 28 T11 treatment 95 81 45 68 T12 treatment 50 60 53 60 P11 placebo 70 52 66 65 P12 placebo 59 68 83 86 T13 treatment 60 61 52 52 T14 treatment 106 52 128 P14 placebo 46 122 131

292 Table S2. Erythropoietin administrated in patients treated with vitamin Eα or placebo at baseline, three months, six months and nine months. Patient VE treatment Baseline 3 months 6 months 9 months code /placebo P1 placebo 37200 16800 23200 33600 T1 treatment 345600 294000 198000 241200 T2 treatment 51600 44400 42000 96000 P2 placebo 252000 271200 324000 295200 P3 placebo 100800 51600 64800 49200 P4 placebo 284400 622800 752400 924000 T3 treatment 19600 151800 78100 278400 T4 treatment 702000 628200 399600 P5 placebo 644400 577200 432000 505200 T5 treatment 276000 115200 124800 192000 T6 treatment 142800 110400 90000 120500 P6 placebo 160800 120000 120000 177600 P7 placebo 104400 37200 32400 27600 P8 placebo 175200 87600 45600 36000 T7 treatment T8 treatment 165600 147600 60000 68400 T9 treatment 250800 306000 385200 397200 P9 placebo 468000 518400 367200 368400 P10 placebo T10 treatment 993600 993600 993600 1009200 T11 treatment 159600 241200 406800 334800 T12 treatment 86400 68400 97200 112800 P11 placebo 18000 8400 17200 75600 P12 placebo 50400 25200 8000 22000 T13 treatment 109200 90000 90000 79200 T14 treatment 20400 60000 26100 P14 placebo 98400 139800 91200

293 Table S3. Ferritin levels of patients treated with vitamin Eα or placebo at baseline, three months, six months and nine months. Patient VE treatment Baseline 3 months 6 months 9 months code /placebo P1 placebo 405 506 469 345 T1 treatment 92 105 205 107 T2 treatment 809 525 386 2271 P2 placebo 523 94 159 280 P3 placebo 414 265 481 P4 placebo 245 189 92 124 T3 treatment 837 287 837 306 T4 treatment 748 610 793 P5 placebo 493 729 793 212 T5 treatment 1092 1543 617 536 T6 treatment 738 1131 536 841 P6 placebo 563 553 774 379 P7 placebo 256 406 661 794 P8 placebo 1268 487 408 273 T7 treatment 215 403 377 T8 treatment 517 1077 424 289 T9 treatment 417 299 165 558 P9 placebo 70 190 171 191 P10 placebo 464 693 877 822 T10 treatment 820 742 706 1029 T11 treatment 740 587 310 580 T12 treatment 850 303 314 633 P11 placebo 598 694 741 745 P12 placebo 212 240 435 396 T13 treatment 500 407 360 462 T14 treatment 796 789 737 P14 placebo 167 459 653

294

O OH

C5H11 CO2Et

1 Figure S50. H NMR (300 MHz, CDCl3) spectrum of (7.1).

O OH

C5H11 CO2Et

13 Figure S51. C NMR (75 MHz, CDCl3) spectrum of (7.1).

295 CO2Et

N C5H11 N H

1 Figure S52. H NMR (300 MHz, CDCl3) spectrum of (7.2).

CO2Et

N C5H11 N H

13 Figure S53. C NMR (75 MHz, CDCl3) spectrum of (7.2).

296 I-(CH2)4CH=CH2

1 Figure S54. H NMR (300 MHz, CDCl3) spectrum of (7.3).

COOH

N C5H11 N (CH2)4CH=CH2

1 Figure S55. H NMR (300 MHz, CDCl3) spectrum of (7.4).

297

COOH

N C5H11 N (CH2)4CH=CH2

13 Figure S56. C NMR (75 MHz, CDCl3) spectrum of (7.4).

N C5H11 N (CH2)4CH=CH2

1 Figure S57. H NMR (400 MHz, CDCl3) spectrum of (7.5).

298 N C5H11 N (CH2)4CH=CH2

13 Figure S58. C NMR (100 MHz, CDCl3) spectrum of (7.5).

COOH

N C5H11 N (CH2)4CH=CH2

Figure S59. HMQC of 3-carboxyl-5-pentyl pyrazole (7.4).

299 COOH

N C5H11 N (CH2)4CH=CH2

Figure S60. HMBC of 3-carboxyl-5-pentyl pyrazole (7.4). O OH

C H C H 5 11 H

1 Figure S61. H NMR (300 MHz, CDCl3) spectrum of (7.7).

300 Br-(CH2)4CH=CH2

1 Figure S62. H NMR (300 MHz, CDCl3) spectrum of (7.8).

NH2-NH-(CH2)4CH=CH2

1 Figure S63. H NMR (300 MHz, CDCl3) spectrum of (7.9).

301 C5H11

N N

(CH2)4CH=CH2

1 Figure S64. H NMR (400 MHz, CDCl3) spectrum of (7.10).

302

Bibliography

303 Aderem, A. A.; Cohen, D. S.; Wright, S. D.; Cohn, Z. A., Bacterial lipopolysaccharides prime macrophages for enhanced release of arachidonic acid metabolites. J. Exp. Med. 1986, 164, (1), 165-79.

Agalou, S.; Ahmed, N.; Babaei-Jadidi, R.; Dawnay, A.; Thornalley, P., Profound mishandling of protein glycation degradation products in uremia and dialysis. J Am Soc Nephrol. 2005, 16, (5), 1471-1485.

Alderson, N. L.; Chachich, M. E.; Frizzell, N.; Canning, P.; Metz, T. O.; Januszewski, A. S.; Youssef, N. N.; Stitt, A. W.; Baynes, J. W.; Thorpe, S. R., Effect of antioxidants and ACE inhibition on chemical modification of proteins and progression of nephropathy in the streptozotocin diabetic rat. . Diabetologia 2004, 47, 1385-1395.

Alderson, N. L.; Chachich, M. E.; Youssef, N. N.; Beattie, R. J.; Nachtigal, M.; Thorpe, S. R.; Baynes, J. W., The AGE inhibitor pyridoxamine inhibits lipemia and development of renal and vascular disease in Zucker obese rats. Kidney Int. 2003, 63, (6), 2123-33.

Alvarez, R. A.; Aguirre, G. D.; Acland, G. M.; Anderson, R. E., Docosapentaenoic acid is converted to docosahexaenoic acid in the retinas of normal and prcd-affected miniature poodle dogs. Invest Ophthalmol Vis Sci 1994, 35, (2), 402-8.

Amarnath, V.; Amarnath, K.; Amarnath, K.; Davies, S.; Roberts, L. J., 2nd, Pyridoxamine: an extremely potent scavenger of 1,4-dicarbonyls. Chem. Res. Toxicol. 2004, 17, (3), 410-5.

Amarnath, V.; Amarnath, K.; Valentine, W. M.; Eng, M. A.; Graham, D. G., Intermediates in the Paal-Knorr synthesis of pyrroles. 4-Oxoaldehydes. Chem. Res. Toxicol. 1995, 8, (2), 234-8.

Anthony, D. C.; Amarnath, V.; Graham, D. G., Pyrrole-mediated protein cross-linking: Toward an understanding of the molecular mechanism. Toxicologist 1990, 10, 183.

Aulak, K. S.; Miyagi, M.; Yan, L.; West, K. A.; Massillon, D.; Crabb, J. W.; Stuehr, D. J., Proteomic method identifies proteins nitrated in vivo during inflammatory challenge. Proc. Natl. Acad. Sci. U. S. A. 2001, 98, (21), 12056-61.

Bacot, S.; Bernoud-Hubac, N.; Baddas, N.; Chantegrel, B.; Deshayes, C.; Doutheau, A.; Lagarde, M.; Guichardant, M., Covalent binding of hydroxy-alkenals 4-HDDE, 4-HHE, and 4-HNE to ethanolamine phospholipid subclasses. J. Lip. Res. 2003, 44, (5), 917-926.

Baragetti, I.; Furiani, S.; Vettoretti, S.; Raselli, S.; Maggi, F. M.; Galli, F.; Catapano, A.

304 L.; Buccianti, G., Role of Vitamin E-Coated Membrane in Reducing Advanced Glycation End Products in Hemodialysis Patients: A Pilot Study. Blood Purif 2006, 24, (4), 369-376.

Barja, G.; Cadenas, S.; Rojas, C.; Perez-Campo, R.; Lopez-Torres, M., Low mitochondrial free radical production per unit O2 consumption can explain the simultaneous presence of high longevity and high aerobic metabolic rate in birds. Free Radic Res 1994, 21, (5), 317-27.

Barton, D. H. R., The invention of chemical reactions of relevance to the chemistry of natural products. Pure & Appl. Chem. 1994, 66, 1943-1954.

Barton, D. H. R.; Crich, D.; Motherwell, W. B., The invention of new radical chain reactions. Part VIII. Radical chemistry of thiohydroxamic esters; A new method for the generation of carbon radicals from carboxylic acids. Tetrahedron 1985, 41, 3901-3924.

Benedetti, A.; Comporti, M.; & Esterbauer, H., Biochim. Biophys. Acta 1980, 620, 281-296.

Benedetti, A.; Comporti, M.; Esterbauer, H., Identification of 4-hydroxynonenal as a cytotoxic product originating from the peroxidation of liver microsomal lipids. Biochim. Biophys. Acta 1980, 620, (2), 281-96.

Ben-Shabat, S.; Itagaki, Y.; Jockusch, S.; Sparrow, J. R.; Turro, N. J.; Nakanishi, K., Formation of a nonaoxirane from A2E, a lipofuscin fluorophore related to macular degeneration, and evidence of singlet oxygen involvement. Angew Chem Int Ed Engl 2002, 41, (5), 814-7.

Bernard, A.; Cocco, M. T.; Maccioni, A.; Plumitallo, A., Phytotoxic activity in pyrazole derivatives. Farmaco 1985, 40, 259-271.

Bernoud-Hubac, N.; Fay, L. B.; Armarnath, V.; Guichardant, M.; Bacot, S.; Davies, S. S.; Jackson Roberts II, L.; Lagarde, M., Covalent binding of isoketals to ethanolamine phosphalipids. Free Radic. Biol. Med. 2004, 37, 1604-1611.

Bicknell, I. R.; Darrow, R.; Barsalou, L.; Fliesler, S. J.; Organisciak, D. T., Mol. Vis. 2002, 8, 333-340.

Bleasdale, C.; Golding, B. T.; Kennedy, G.; MacGregor, J. O.; Watson, W. P., Reactions of muconaldehyde isomers with nucleophiles including tri-O-acetylguanosine: formation of 1,2-disubstituted pyrroles from reactions of the (Z,Z)-isomer with primary amines.

305 Chem. Res. Toxicol. 1993, 6, (4), 407-12.

Bligh, E.; and Dyer, W., A rapid method of total lipid extraction and purification. Can. J. Biochem. Physiol. 1956, 37, 911-917.

Boaz, M.; Smetana, S.; Weinstein, T.; Matas, Z.; Gafter, U.; Iaina, A.; Knecht, A.; Weissgarten, Y.; Brunner, D.; Fainaru, M.; Green, M., Secondary prevention with antioxidants of cardiovascular disease in endstage renal disease (SPACE): randomised placebo-controlled trial. Lancet 2000, 356, 1213-1218.

Bok, D., New insights and new approaches toward the study of age-related macular degeneration. Proc. Natl. Acad. Sci. U. S. A. 2002, 99, 14619-14621.

Bolland, J. L., Kinetics of olefin oxidation. Q. Rev. Chem. Soc. 1949, 3, 1.

Booth, A. A.; Khalifah, R. G.; Todd, P.; Hudson, B. G., In vitro kinetic studies of formation of antigenic advanced glycation end products (AGEs). Novel inhibition of post-Amadori glycation pathways. J. Biol. Chem. 1997, 272, (9), 5430-7.

Borgeat, P.; Samuelsson, B., Arachidonic acid metabolism in polymorphonuclear leukocytes: effects of ionophore A23187. Proc. Natl. Acad. Sci. U. S. A. 1979, 76, (5), 2148-52.

Borgeat, P.; Samuelsson, B., Transformation of arachidonic acid by rabbit polymorphonuclear leukocytes. Formation of a novel dihydroxyeicosatetraenoic acid. J. Biol. Chem. 1979, 254, (8), 2643-6.

Bottoms, G. D.; Johnson, M. A.; Lamar, C. H.; Fessler, J. F.; Turek, J. J., Endotoxin-induced eicosanoid production by equine vascular endothelial cells and neutrophils. Circ Shock 1985, 15, (3), 155-62.

Boulton, M.; Rozanowska, M.; Rozanowski, B., J. Photochem. Pathobiol. B: Biology 2001, 64.

Boutaud, O.; Brame, C. J.; Salomon, R. G.; Roberts, L. J., 2nd; Oates, J. A., Characterization of the lysyl adducts formed from prostaglandin H2 via the levuglandin pathway. Biochemistry 1999, 38, (29), 9389-96.

Brame, C. J.; Salomon, R. G.; Morrow, J. D.; Roberts, L. J., 2nd, Identification of extremely reactive gamma-ketoaldehydes (isolevuglandins) as products of the isoprostane pathway and characterization of their lysyl protein adducts. J. Biol. Chem. 1999, 274,

306 (19), 13139-46.

Brennan, M. L.; Wu, W. J.; Fu, X. M.; Shen, Z. Z.; Song, W.; Frost, H.; Vadseth, C.; Narine, L.; Lenkiewicz, E.; Borchers, M. T.; Lusis, A. J.; Lee, J. J.; Lee, N. A.; Abu-Soud, H. M.; Ischiropoulos, H.; Hazen, S. L., A tale of two controversies - Defining both the role of peroxidases in nitrotyrosine formation in vivo using eosinophil peroxidase and myeloperoxidase-deficient mice, and the nature of peroxidase-generated reactive nitrogen species. J. Biol. Chem. 2002, 277, (20), 17415-17427.

Brown, B. G.; Crowley, J., Is there any hope for vitamin E? JAMA 2005, 293, (11), 1387-90.

Brown, B. G.; Zhao, X. Q.; Chait, A.; Fisher, L. D.; Cheung, M. C.; Morse, J. S.; Dowdy, A. A.; Marino, E. K.; Bolson, E. L.; Alaupovic, P.; Frohlich, J.; Albers, J. J., Simvastatin and niacin, antioxidant vitamins, or the combination for the prevention of coronary disease. N Engl J Med 2001, 345, (22), 1583-92.

Bryson, S. E.; Smith, I. M., Epidemiology of autism: prevalence, associated characteristics, and implications for research and service delivery. . Ment Retard Dev Disabil Res Rev 1998, 4, 97-103.

Bucala, R.; Makita, Z.; Koschinsky, T.; Cerami, A.; Vlassara, H., Lipid Advanced Glycosylation - Pathway for Lipid Oxidation in-Vivo. Proc. Natl. Acad. Sci. USA 1993, 90, (14), 6434-6438.

Buettner, G., The pecking order of free radicals and antioxidants: lipid peroxidation, alpha-tocopherol and ascorbate. Arch. Biochem. Biophys. 1993, 300, 535-543.

Burton, G. W. a. I., K. U., Autoxidation of biological molecules. 1. The antioxidant activity of vitamin e and related chain-breaking phenolic antioxidants in vitro. J. Am. Chem. Soc. 1981, 103, 6472-6477.

Cadenas, E.; Muller, A.; Brigelius, R.; Esterbauer, H.; Sies, H., Biochem. J. 1983, 214, 479-487.

Carlson, E. C.; Drazba, J.; Yang, X.; Perez, V. L., Visualization and characterization of inflammatory cell recruitment and migration through the corneal stroma in endotoxin-induced keratitis. Invest Ophthalmol Vis Sci 2006, 47, (1), 241-8.

Cason, J.; Chang, M. P., Condensation of acrylonitrile with unsymmetrical ketones. J. Org. Chem. 1956, 21, 449-454.

307

Chen, J. J.; Yu, B. P., Alterations in mitochondrial membrane fluidity by lipid peroxidation products. Free Radic Biol Med 1994, 17, (5), 411-8.

Chen, P.; Wiesler, D.; Chmelı´k, J.; Novotny, M., Substituted 2-Hydroxy-1,2-dihydropyrrol-3-ones: Fluorescent Markers Pertaining to Oxidative Stress and Aging. Chem. Res. Toxicol. 1996, 9, 970-979.

Chetty, C.; Klapper, D. G.; Schwab, J. H., Soluble peptidoglycan-polysaccharide fragments of the bacterial cell wall induce acute inflammation. Infect. Immun. 1982, 38, (3), 1010-9.

Chio, K. S.; Tappel, A. L., Synthesis and characterization of the fluorescent products derived from malonaldehyde and amino acids. Biochemistry 1969, 8, (7), 2821-6.

Christen, S.; Woodall, A.; Shigenaga, M.; Southwell-Keely, P.; Duncan, M.; Ames, B.,

-tocopherol traps mutagenic electrophiles such as NOx and complements -tocopherol:Physiological implications. Proc Natl Acad Sci USA 1997, 94, 3217-3222.

Clark, W. R., A means to an end : the biological basis of aging and death. 1999.

Comi, A. M.; Zimmerman, A. W.; Frye, V. H.; Law, P. A.; Peeden, J. N., Familial clustering of autoimmune disorders and evaluation of medical risk factors in autism. J. Child Neurol. 1999, 14, 388-394.

Connolly, A. M.; Chez, M. G.; Pestronk, A.; Arnold, S. T.; Mehta, S.; Deuel, R. K., Serum autoantibodies to brain in Landau-Kleffner variant, autism, and other neurologic disorders. J. Pediatr. 1999, 134, 607-613.

Crabb, J. W.; Miyagi, M.; Gu, X.; Shadrach, K.; West, K. A.; Sakaguchi, H.; Kamei, M.; Hasan, A.; Yan, L.; Rayborn, M. E.; Salomon, R. G.; Hollyfield, J. G., Drusen proteome analysis: an approach to the etiology of age-related macular degeneration. Proc. Natl. Acad. Sci. USA 2002, 99, (23), 14682-7.

Crabb, J. W.; Miyagi, M.; Gu, X. R.; Shadrach, K.; West, K. A.; Sakaguchi, H.; Kamei, M.; Hasan, A.; Yan, L.; Rayborn, M. E.; Salomon, R. G.; Hollyfield, J. G., Drusen proteome analysis: An approach to the etiology of age-related macular degeneration. Proc. Natl. Acad. Sci. U. S. A. 2002, 99, (23), 14682-14687.

Crabb, J. W.; Nie, Z.; Chen, Y.; Hulmes, J. D.; West, K. A.; Kapron, J. T.; Ruuska, S. E.; Noy, N.; Saari, J. C., Cellular retinaldehyde-binding protein ligand interactions. Gln-210

308 and Lys-221 are in the retinoid binding pocket. J. Biol. Chem. 1998, 273, (33), 20712-20.

Criegee, R.; Pilz, H.; Flygare, H., Chem. Ber. 1939, 72, 1799.

Cristol, J. P.; Canaud, B.; Rabesandratana, H.; Gaillard, I.; Serre, A.; Mion, C., Enhancement of reactive oxygen species production and cell surface markers expression due to haemodialysis. Nephrol Dial Transplant 1994, 9, (4), 389-94.

Davies, M. J.; Fu, S.; Dean, R. T., Protein hydroperoxides can give rise to reactive free radicals. Biochem. J. 1995, 305, 643-649.

Davies, S. S.; Amarnath, V.; Montine, K. S.; Bernoud-Hubac, N.; Boutaud, O.; Montine, T. J.; Roberts, L. J., 2nd, Effects of reactive gamma-ketoaldehydes formed by the isoprostane pathway (isoketals) and cyclooxygenase pathway (levuglandins) on proteasome function. FASEB J. 2002, 16, (7), 715-7.

Davies, S. S.; Brantley, E. J.; Voziyan, P. A.; Amarnath, V.; Zagol-Ikapitte, I.; Boutaud, O.; Hudson, B. G.; Oates, J. A.; Ii, L. J., Pyridoxamine Analogues Scavenge Lipid-Derived gamma-Ketoaldehydes and Protect against H(2)O(2)-Mediated Cytotoxicity. Biochemistry 2006, 45, (51), 15756-15767.

Dean, R. T.; Gebicki, J.; Gieseg, S.; Grant, A. J.; Simpson, J. A., Hypothesis: a damaging role in aging for reactive protein oxidation products? Mutat. Res. 1992, 275, 387-393.

Dean, R. T.; Gieseg, S.; Davies, M. J., Reactive species and their accumulation on radical-damaged proteins. Trends Biochem.Sci. 1993, 18, 437-441.

DeCaprio, A. P., Mechanisms of in vitro pyrrole adduct autoxidation in 2,5-hexanedione-treated protein. Mol. Pharmacol. 1986, 30, (5), 452-8.

DeCaprio, A. P.; Jackowshi, S. J.; and Regan, K. A., Mechanism of formation and quantitation of imines, pyrroles, and stable nonpyrrole adducts in 2,5-hexanedione-treated protein. Mol. Pharmacol. 1987, 32, 542-548.

Decaprio, A. P.; Jackowski, S. J.; Regan, K. A., Mechanism of Formation and Quantitation of Imines, Pyrroles, and Stable Nonpyrrole Adducts in 2,5-Hexanedione-Treated Protein. Mol. Pharmacol. 1987, 32, (4), 542-548.

DeCaprio, A. P.; Olajos, E. J.; Weber, P., Covalent binding of a neurotoxic n-hexane metabolite: conversion of primary amines to substituted pyrrole adducts by 2,5-hexanedione. Toxicol. Appl. Pharmacol. 1982, 65, (3), 440-50.

309

Degenhardt, T. P.; Alderson, N. L.; Arrington, D. D.; Beattie, R. J.; Basgen, J. M.; Steffes, M. W.; Thorpe, S. R.; Baynes, J. W.; , Pyridoxamine inhibits early renal disease and dyslipidemia in the streptozotocin-diabetic rat. Kidney Int 2002, 61, 939-950.

Dekkers, J. C.; Van Doornen, L. J.; and Kemper, H. C. G., The Role of Antioxidant Vitamins and Enzymes in the Prevention of Exercise-Induced Muscle Damage. Sports Med 1996, 21, 213-238.

DeMaio, S.; King, S.; Lembo, N.; Roubin, G.; Hearn, J.; Bhagavan, H.; Sgoutas, D., Vitamin E supplementation, plasma lipids and incidence of restenosis after percutaneous transluminal coronary angioplasty (PTCA). J Am Coll Nutr 1992, 11, 68-73.

Deneke, S. M., Thiol-based antioxidants. Curr Top Cell Regul, 2000, 36, 151-180.

Dennis, E. A., Diversity of group types, regulation, and function of phospholipase A2. J. Biol. Chem. 1994, 269, 13057-13060.

Devaraj, S.; Jialal, I., Failure of vitamin E in clinical trials: is gamma-tocopherol the answer? Nutr Rev 2005, 63, (8), 290-3.

Diepeveen, S. H.; Verhoeven, G. W.; Van Der Palen, J.; Dikkeschei, L. D.; Van Tits, L. J.; Kolsters, G.; Offerman, J. J.; Bilo, H. J.; Stalenhoef, A. F., Effects of atorvastatin and vitamin E on lipoproteins and oxidative stress in dialysis patients: a randomised-controlled trial. J Intern Med 2005, 257, (5), 438-45.

DiFranco, E.; Subbanagounder, G.; Kim, S.; Murthi, K.; Taneda, S.; Monnier, V. M.; Salomon, R. G., Formation and stability of pyrrole adducts in the reaction of levuglandin E2 with proteins. Chem. Res. Toxicol. 1995, 8, (1), 61-7.

Dinarello, C. A.; Conti, P.; Mier, J. W., Effects of human interleukin-1 on natural killer cell activity: is fever a host defense mechanism for tumor killing? Yale J Biol Med 1986, 59, (2), 97-106.

Dinarello, C. A.; Mier, J. W., Interleukins. Annu Rev Med 1986, 37, 173-8.

Doerfler, M. E.; Danner, R. L.; Shelhamer, J. H.; Parrillo, J. E., Bacterial lipopolysaccharides prime human neutrophils for enhanced production of leukotriene B4. J. Clin. Invest. 1989, 83, (3), 970-7.

Eapen, K. C.; Tamborski, C., Synthesis of 1,3,5-tri-n-alkylbenzene compounds. J. Org.

310 Chem. 1988, 53, 5564-5567.

Ebrahem, Q.; Renganathan, K.; Sears, J.; Vasanji, A.; Gu, X.; Lu, L.; Salomon, R. G.; Crabb, J. W.; Anand-Apte, B., Carboxyethylpyrrole oxidative protein modifications stimulate neovascularization: Implications for age-related macular degeneration. Proc. Natl. Acad. Sci. USA 2006, 103, (36), 13480-4.

Ebrahem, Q.; Renganathan, K.; Sears, J.; Vasanji, A.; Gu, X. R.; Lu, L.; Salomon, R. G.; Crabb, J. W.; Anand-Apte, B., Carboxyethylpyrrole oxidative protein modifications stimulate neovascularization: Implications for age-related macular degeneration. Proc. Natl. Acad. Sci. U. S. A. 2006, 103, (36), 13480-13484.

Economides, P. A.; Khaodhiar, L.; Caselli, A.; Caballero, A. E.; Keenan, H.; Bursell, S. E.; King, G. L.; Johnstone, M. T.; Horton, E. S.; Veves, A., The effect of vitamin E on endothelial function of micro- and macrocirculation and left ventricular function in type 1 and type 2 diabetic patients. Diabetes 2005, 54, (1), 204-11.

Edelson, S. M., Overview of autism. http://www.autism.org/overview.html.

Elguero, J., Comprehensive heterocyclic chemistry. Pergamon Press: Oxford. 1996, 3, 1-75.

Esser, R. E.; Anderle, S. K.; Chetty, C.; Stimpson, S. A.; Cromartie, W. J.; Schwab, J. H., Comparison of inflammatory reactions induced by intraarticular injection of bacterial cell wall polymers. Am. J. Pathol. 1986, 122, (2), 323-34.

Esterbauer, H.; Koller, E.; Slee, R. G.; Koster, J. F., Possible Involvement of the Lipid-Peroxidation Product 4-Hydroxynonenal in the Formation of Fluorescent Chromolipids. Biochem. J. 1986, 239, (2), 405-409.

Esterbauer, H.; Schaur, R. J.; Zollner, H., Chemistry and biochemistry of 4-hydroxynonenal, malonaldehyde and related aldehydes. Free Radic Biol Med 1991, 11, (1), 81-128.

Esterbauer, H.; Zollner, H.; Lang, J., Metabolism of the lipid peroxidation product 4-hydroxynonenal by isolated hepatocytes and by liver cytosolic fractions. Biochem. J. 1985, 228, (2), 363-73.

Evans, T.; Siedlak, S.; Lu, L.; Fu, X.; Wang, Z.; McGinnis, W.; Fakhoury, E.; Castellani, R.; Hazen, S. L.; Walsh, W. J.; Salomon, R. G.; Smith, M. A.; Perry, G.; Zhu, X., The Autistic Phenotype Exhibits a Remarkably Localized Modification of Brain Protein by

311 Products of Free Radical-Induced Lipid Oxidation. Am. J. Biochem. Biotechnol. 2007, In press.

Farnsworth, C. C.; Dratz, E. A., Oxidative damage of retinal rod outer segment membranes and the role of vitamin E. Biochim. Biophys. Acta 1976, 443, (3), 556-70.

Ferris, F. L., 3rd American Journal of Epidemiology 1983, 118, 132-151.

Finkel, T., Oxygen radicals and signaling. Curr. Opin. Cell Biol. 1998, 10, (2), 248-53.

Finkel, T.; Holbrook, N. J., Oxidants, oxidative stress and the biology of ageing. Nature 2000, 408, (6809), 239-47.

Fliesler, S. J.; Anderson, R. E., Chemistry and Metabolism of Lipids in the Vertebrate Retina. Prog. Lipid. Res. 1983, 22, (2), 79-131.

Ford, E. S.; Ajani, U. A.; Mokdad, A. H., Brief communication: The prevalence of high intake of vitamin E from the use of supplements among U.S. adults. Ann. Intern. Med. 2005, 143, (2), 116-20.

Freedman, J. H.; Ciriolo, M. R.; Peisach, J., The role of glutathione in copper metabolism and toxicity. J. Biol. Chem. 1989, 264, 5598-5605.

Friedlander, M.; Witko-Sarsat, V.; Nguyen, A.; Wu, Y.; Labrunie, M.; Verger, C.; Jungers, P.; Descamps-Latscha, B., The advanced glycation endproduct pentosidine and monocyte activation in uremia. Clin Nephrol 1996, 45, 379-382.

Friedlander, M.; Wu, Y.; Elgawish, A.; Monnier, V., Early and Advanced Glycosylation End Products: Kinetics of formation and clearance in peritoneal dialysis. J. Clin. Invest 1996, 97, 728-735.

Friedlander, M.; Wu, Y.; Schulak, J.; Monnier, V.; Hricik, D., Influence of dialysis modality on plasma and tissue concentrations of pentosidine in patients with end-stage renal disease. Am J Kidney Dis 1995, 25, (3), 445-451.

Fu, J. Y.; Masferrer, J. L.; Seibert, K.; Raz, A.; Needleman, P., The induction and suppression of prostaglandin H2 synthase (cyclooxygenase) in human monocytes. J. Biol. Chem. 1990, 265, (28), 16737-40.

Gaillard, E. R.; Atherton, S. J.; Eldred, G.; Dillon, J., Photophysical studies on human retinal lipofuscin. Photochem. Photobiol. 1995, 61, (5), 448-53.

312

Galli, F.; Benedetti, S.; Floridi, A.; Canestrari, F.; Piroddi, M.; Buoncristiani, E.; Buoncristiani, U., Glycoxidation and inflammatory markers in patients on treatment with PMMA-based protein-leaking dialyzers. Kidney Int. 2005, 67, (2), 750-9.

Galli, F.; Buoncristiani, U.; Conte, C.; Aisa, C.; Floridi, A., Vitamin E in uremia and dialysis patients. Annals NY Acad Sci 2004, 1031, 348-351.

Gardini, G. P., The oxidation of monocyclic pyrroles. Adv. Heterocycl. Chem. 1973, 15, 67-99.

Ghiadoni, L.; Cupisti, A.; Huang, Y.; Mattei, P.; Cardinal, H.; Favilla, S.; Rindi, P.; Barsotti, G.; Taddei, S.; Salvetti, A., Endothelial dysfunction and oxidative stress in chronic renal failure. J Nephrol 2004, 17, (4), 512-9.

Girndt, M.; Lengler, S.; Kaul, H.; Sester, U.; Sester, M.; Kohler, H., Prospective crossover trial of the influence of vitamin-E coated dialyzer membranes on T-cell activation and cytokine induction. Am J Kidney Dis 2000, 35, 95-104.

Golse, B.; Debray-Ritzen, P.; Durosay, P.; Puget, K.; Michelson, A. M., Alterations in two enzymes: superoxide dismutase and glutathion peroxidase in developmental infantile psychosi (infantile autism) (author's transl). Rev. Neurol. (Paris) 1978, 134, 699-705.

Gossauer, A.; Nesvadba, P., Oxidation and reduction of the pyrrole ring. . Pyrroles Part One: The Synthesis and the Physical Aspects of the Pyrrole Ring (Jones, R. A., Ed.) Wiley, New York. 1990, 499-536.

Graham, D. G.; Anthony, D. C.; Boekelheide, K.; Maschmann, N. A.; Richards, R. G.; Wolfram, J. W.; Shaw, B. R., Studies of the molecular pathogenesis of hexane neuropathy. II. Evidence that pyrrole derivatization of lysyl residues leads to protein crosslinking. Toxicol. Appl. Pharmacol. 1982, 64, (3), 415-22.

Greenberg, M. E.; Sun, M.; Zhang, R.; Febbraio, M.; Silverstein, R.; Hazen, S. L., Oxidized phosphatidylserine-CD36 interactions play an essential role in macrophage-dependent phagocytosis of apoptotic cells. J. Exp. Med. 2006, 203, (12), 2613-25.

Grisham, M. B., Oxidants and free radicals in inflammatory bowel disease. Lancet 1994, 344, (8926), 859-61.

Gu, X.; Meer, S. G.; Miyagi, M.; Rayborn, M. E.; Hollyfield, J. G.; Crabb, J. W.;

313 Salomon, R. G., Carboxyethylpyrrole protein adducts and autoantibodies, biomarkers for age-related macular degeneration. J. Biol. Chem. 2003, 278, 42027-42035.

Gu, X.; Sun, M.; Gugiu, B.; Hazen, S.; Crabb, J. W.; Salomon, R. G., Oxidatively truncated docosahexaenoate phospholipids: total synthesis, generation, and Peptide adduction chemistry. J. Org. Chem. 2003, 68, (10), 3749-61.

Gu, X. R.; Sun, M. J.; Gugiu, B.; Hazen, S.; Crabb, J. W.; Salomon, R. G., Oxidatively truncated docosahexaenoate phospholipids: Total synthesis, generation, and peptide adduction chemistry. J. Org. Chem. 2003, 68, (10), 3749-3761.

Guichardant, M.; Bernoud-Hubac, N.; Chantegrel, B.; Deshayes, C.; Lagarde, M., Aldehydes from n-6 fatty acid peroxidation. Effects on aminophospholipids. Prostaglandins Leukot. Essent. Fatty Acids 2002, 67, (2-3), 147-149.

Gupta, S.; Aggarwal, S.; Heads, C., Dysregulated immune system in children with autism: Beneficial effects of intravenous immune globulin on autistic characteristics. J. Autism Dev. Disord. 1996, 26, 439-452.

Gupta, S.; Aggarwal, S.; Rashanravan, B.; Lee, T., Th1- and Th2-like cytokines in CD4+ and CD8+ T cells in autism. J. Neuroimmunol. 1998, 85, (1), 106-9.

Habraken, C. L.; Moore, J. A., Heterocyclic studies. XVI. The assignment of isomeric and tautomeric structures of pyrazoles by nuclear magnetic resonance. J. Org. Chem. 1965, 30, 1892-1896.

Halliwell, B., The antioxidant paradox. Lancet 2000, 355, 1179-80.

Halliwell, B.; Zhao, K.; and Whiteman, M., The ugly, the uglier and the not so good: a personal view of recent controversies. Free Radic. Res. 1999, 31, 651-669.

Hamberg, M.; Samuelsson, B., Detection and isolation of an endoperoxide intermediate in prostaglandin biosynthesis. Proc. Natl. Acad. Sci. U. S. A. 1973, 70, (3), 899-903.

Hamberg, M.; Samuelsson, B., Prostaglandin endoperoxides. Novel transformations of arachidonic acid in human platelets. Proc. Natl. Acad. Sci. U. S. A. 1974, 71, (9), 3400-4.

Handelman, G.; Walter, M.; Adhikarla, R.; Gross, J.; Dallai, G.; Lewin, N., Elevated plasma F2-isoprostanes in patients on long-term hemodialysis. Kidney Int. 2001, 59, 1960-1966.

314 Handelman, G. J.; Epstein, W. L.; Peerson, J.; Spiegelman, D.; Machlin, L. J.; Dratz, E. A., Human adipose alpha-tocopherol and gamma-tocopherol kinetics during and after 1 y of alpha-tocopherol supplementation. Am. J. Clin. Nutr. 1994, 59, (5), 1025-32.

Handelman, G. J.; Machlin, L. J.; Fitch, K.; Weiter, J. J.; Dratz, E. A., Oral alpha-tocopherol supplements decrease plasma gamma-tocopherol levels in humans. J. Nutr. 1985, 115, (6), 807-13.

Harman, D., Aging: A theory based on free radical and radiation chemistry. J. Gerontol. 1956, 2, 298-300.

Harman, D., The aging process. Proc. Natl. Acad. Sci. USA 1981, 78, 7124-7128.

Harris, L. K.; Mann, G. E.; Ruiz, E.; Mushtaq, S.; Leake, D. S., Ascorbate does not protect macrophages against apoptosis induced by oxidised low density lipoprotein. Arch. Biochem. Biophys. 2006, 455, (1), 68-76.

Harris, T. M.; Boatman, S.; Hauser, C. R., Alkylations at the position of acetoacetaldehyde and benzylacetoacetaldehyde through their dicarbanions. J. Am. Chem. Soc. 1963, 85, 3273-3276.

Hayase, F.; Nagaraj, R. H.; Miyata, S.; MNjoroge, F. G.; Monnier, V. M., J. Biol. Chem. 1989, 264, 3758.

Heller, J. I.; Crowley, J. R.; Hazen, S. L.; Salvay, D. M.; Wagner, P.; Pennathur, S.; Heinecke, J. W., p-Hydroxyphenylacetaldehyde, an aldehyde generated by myeloperoxidase, modifies phospholipid amino groups of low density lipoprotein in human atherosclerotic intima. J. Biol. Chem. 2000, 275, (14), 9957-9962.

Himmelfarb, J.; Hakim, R. M., Oxidative stress in uremia. Curr Opin Nephrol Hypertens 2003, 12, (6), 593-8.

Himmelfarb, J.; Kane, J.; McMonagle, E.; Zaltas, E.; Bobzin, S.; Boddupalli, S.; Phinney, S.; Miller, G., Alpha and gamma tocopherol metabolism in healthy subjects and patients with end-stage renal disease. Kidney Int. 2003, 64, 978-991.

Himmelfarb, J.; Stenvinkel, P.; Ikizler, T. A.; Hakim, R. M., The elephant in uremia: oxidant stress as a unifying concept of cardiovascular disease in uremia. Kidney Int. 2002, 62, (5), 1524-38.

Hla, T.; Neilson, K., Human cyclooxygenase-2 cDNA. Proc. Natl. Acad. Sci. U. S. A.

315 1992, 89, (16), 7384-8.

Hoff, H. F.; O'Neil, J.; Wu, Z.; Hoppe, G.; Salomon, R. L., Phospholipid hydroxyalkenals: biological and chemical properties of specific oxidized lipids present in atherosclerotic lesions. Arterioscler Thromb Vasc Biol 2003, 23, (2), 275-82.

Hoft, E.; Katritzky, A. R.; Nesbit, M. R., The autoxidation of alkylpyrroles. Tetrahedron Lett. 1967, 32, 3041-3044.

Hollyfield, J. G.; Rayborn, M. E.; Yang, X.; Ufret, R.; Lu, L.; Yu, M.; Shadrach, K. G.; Peachey, N. S.; Salomon, R. G.; Perez, V. L., Identification of an Inflammatory Signal From the Outer Retina Causing Age-Related Macular Degeneration. 2007 ARVO Annual Meeting, May 6-10, 2007, Fort Lauderdale, FL, U. S. A.

Holz, F. G.; Schutt, F.; Kopitz, J.; Eldred, G. E.; Kruse, F. E.; Volcker, H. E.; Cantz, M., Inhibition of lysosomal degradative functions in RPE cells by a retinoid component of lipofuscin. Invest. Ophthalmol. Vis. Sci. 1999, 40, (3), 737-43.

Holz, F. G.; Schutt, F.; Kopitz, J.; Volcker, H. E., Coupling of the lipofuscin fluorophor (A2-E) to LDL particles allows specific loading of the lysosomal compartment in cultured human RPE cells. Ophthalmologe. 1999, 96, (12), 781-785.

Horkko, S.; Miller, E.; Dudl, E.; Reaven, P.; Curtiss, L. K.; Zvaifler, N. J.; Terkeltaub, R.; Pierangeli, S. S.; Branch, D. W.; Palinski, W.; Witztum, J. L., Antiphospholipid antibodies are directed against epitopes of oxidized phospholipids. Recognition of cardiolipin by monoclonal antibodies to epitopes of oxidized low density lipoprotein. J. Clin. Invest. 1996, 98, (3), 815-25.

Horrocks L. A.; Yeo, Y. K., Health Benefits Of Docosahexaenoic Acid (DHA). Pharmacol. Res. 1999, 40, (3), 211-225.

Hosomi, A.; Arita, M.; Sato, Y.; Kiyose, C.; Ueda, T.; Igarashi, O.; Arai, H.; Inoue, K., Affinity for alpha-tocopherol transfer protein as a determinant of the biological activities of vitamin E analogs. FEBS Lett. 1997, 409, (1), 105-8.

Hunter, D. J.; Manson, J. E.; Colditz, G. A.; Stampfer, M. J.; Rosner, B.; Hennekens, C. H.; Speizer, F. E.; Willett, W. C., A prospective study of the intake of vitamins C, E, and A and the risk of breast cancer. N Engl J Med 1993, 329, (4), 234-40.

Hviid, A.; Stellfeld, M.; Wohlfahrt, J.; Melbye, M., Association between thimerosal-containing vaccine and autism. JAMA 2003, 290, 1763-1766.

316

Ikizler, T.; Moroz, J.; Roberts, L.; Evanson, J.; Becker, B.; Hakim, R.; Shire, Y.; Himmelfarb, J., Plasma F2-isprostanes levels are elevated in chronic hemodialysis patients. Clin Nephrol 2002, 58, 190-197.

Islam, K. N.; O'Byrne, D.; Devaraj, S.; Palmer, B.; Grundy, S. M.; Jialal, I., Alpha-tocopherol supplementation decreases the oxidative susceptibility of LDL in renal failure patients on dialysis therapy. Atherosclerosis 2000, 150, (1), 217-24.

Iyer, R. S.; Ghosh, S.; Salomon, R. G., Levuglandin E2 crosslinks proteins. Prostaglandins 1989, 37, (4), 471-80.

Jackson, A. H., Pyrroles. . Comprehensive Organic Chemistry (Barton, D., and Ollis, D. W., Eds.) 1980, 4, pp 275-320, peergamon, New York.

Jain, S. K., The accumulation of malonyldialdehyde, a product of fatty acid peroxidation, can disturb aminophospholipid organization in the membrane bilayer of human erythrocytes. J. Biol. Chem. 1984, 259, (6), 3391-4.

James, S. J.; Cutler, P.; Melnyk, S.; Hernigan, S.; Janak, L.; Gaylor, D. W.; Neubrander, J. A., Metabolic biomarkers of increased oxidative stress and methylation capacity in children with autism. . Am. J. Clin. Nutr. 2004, 80, 1611-1617.

Jang, Y. P.; Matsuda, H.; Itagaki, Y.; Nakanishi, K.; Sparrow, J. R., Characterization of peroxy-A2E and furan-A2E photooxidation products and detection in human and mouse retinal pigment epithelial cell lipofuscin. J. Biol. Chem. 2005, 280, (48), 39732-9.

Jiang, J.; St Croix, C. M.; Sussman, N.; Zhao, Q.; Pitt, B. R.; Kagan, V. E., Contribution of glutathione and metallothioneins to protection against copper toxicity and redox cycling: quantitative analysis using MT+/+ and MT-/- mouse lung fibroblast cells. Chem. Res. Toxicol. 2002, 15, 1080-1087.

Jiang, Q.; Ames, B. N., Gamma-tocopherol, but not alpha-tocopherol, decreases proinflammatory eicosanoids and inflammation damage in rats. FASEB J. 2003, 17, (8), 816-22.

Jiang, Q.; Christen, S.; Shigenaga, M. K.; Ames, B. N., gamma-tocopherol, the major form of vitamin E in the US diet, deserves more attention. Am. J. Clin. Nutr. 2001, 74, (6), 714-22.

Jiang, Q.; Elson-Schwab, I.; Courtemanche, C.; Ames, B. N., gamma-tocopherol and its

317 major metabolite, in contrast to alpha-tocopherol, inhibit cyclooxygenase activity in macrophages and epithelial cells. Proc. Natl. Acad. Sci. U. S. A. 2000, 97, (21), 11494-9.

Jiang, Q.; Wong, J.; Fyrst, H.; Saba, J. D.; Ames, B. N., gamma-Tocopherol or combinations of vitamin E forms induce cell death in human prostate cancer cells by interrupting sphingolipid synthesis. Proc. Natl. Acad. Sci. U. S. A. 2004, 101, (51), 17825-30.

Jones, G. B.; Chapman, B. J., Decarboxylation of indole-2-carboxylic acids: Improved procedures. . J. Org. Chem. 1993, 58, 5558-5559.

Jyonouchi H; Sun S; H, L., Proinflammatory and regulatory cytokine production associated with innate and adaptive immune responses in children with autism spectrum disorders and developmental regression. J. Neuroimmunol. 2001, 120, 170-179.

Kaczmarski, M., J. Wojicicki, L. Samochowiee, T. Dutkiewicz, and Z. Sych., The influence of exogenous antioxidants and physical exercise on some parameters associated with production and removal of free radicals. Pharmazie. 1999, 54, 303-306.

Kalyanaraman, B.; and Gutterman, D. D., Prologue: Vascular effects of free radicals Am J Physiol Heart Circ Physiol 2003, 285, H2253-H2254.

Kanner, L., Autistic disturbances of affective contact. Nervous Child 1943, 2, 217-250.

Kano, K.; Scarpetti, D.; Warner, J. C.; Anselme, J. P.; Springer, J. P.; Arison, B. H., Benzoylphenyl-1-methylpyrazoles. Can. J. Chem. 1986, 64, 2211-2219.

Katritzky, A. R.; Nesbit, M. R., The autoxidation of alkylpyrroles. Tetrahedron Lett. 1967, 32, 3041-3044.

Kaur, K.; Salomon, R. G.; O'Neil, J.; Hoff, H. F., (Carboxyalkyl)pyrroles in human plasma and oxidized low-density lipoproteins. Chem. Res. Toxicol. 1997, 10, (12), 1387-1396.

Kaushal, R.; Sorani, S.; Deshpande, S. S., J. Ind. Chem. Soc. 1942, 19, 107.

Khalifah, R. G.; Todd, P.; Booth, A. A.; Yang, S. X.; Mott, J. D.; Hudson, B. G., Kinetics of nonenzymatic glycation of ribonuclease A leading to advanced glycation end products. Paradoxical inhibition by ribose leads to facile isolation of protein intermediate for rapid post-Amadori studies. Biochemistry 1996, 35, (15), 4645-54.

318 Kidd, P. M., Autism, an extreme challenge to integrative medicine. Part 1: the knowledge base. Alternative Medicine Review. 2002, http://www.findarticles.com/p/articles/mi_m0FDN/is_4_7/ai_91155402.

Kikugawa, K.; Kato, T.; Beppu, M.; Hayasaka, A., Fluorescent and cross-linked proteins formed by free radical and aldehyde species generated during lipid oxidation. Adv. Exp. Med. Biol. 1989, 266, 345-356; discussion 357.

King, C. M.; Bristow-craig, H. E.; Gillespie, E. S.; Barnett, Y. A., In vivo antioxidant status, DNA damage, mutation and DNA repair capacity in cultured lymphocytes from healthy 75- to8 0-year-old humans. Mutat. Res. 1997, 377, 137-147.

Klein, R.; Wang, Q.; Klein, B. E.; Moss, S. E.; Meuer, S. M., The relationship of age-related maculopathy, cataract, and glaucoma to visual acuity. Invest Ophthalmol Vis Sci 1995, 36, (1), 182-91.

Knight, J. A., Review: Free radicals, antioxidants, and the immune system Ann. Clini. Lab. Sci. 2000, 30, 145-158.

Kobierski, M. E.; Kim, S.; Murthi, K. K.; Iyer, R. S.; Salomon, R. G., Synthesis of a pyrazole isostere of pyrroles formed by the reaction of the amino groups of protein

lysyl residues with levuglandin E2. J. Org. Chem. 1994, 59, 6044-6050.

Korvatska, E.; Van De Water, J.; Anders, T. F.; Gershwin, M. E., Genetic and immunologic considerations in autism. Neurobiol. Dis. 2002, 9, 107-125.

Kruijff, B., Nature 1997, 386, 129-130.

Krzystolik, M. G.; Afshari, M. A.; Adamis, A. P.; Gaudreault, J.; Gragoudas, E. S.; Michaud, N. A.; Li, W. J.; Connolly, E.; O'Neill, C. A.; Miller, J. W., Prevention of experimental choroidal neovascularization with intravitreal anti-vascular endothelial growth factor antibody fragment. Archives of Ophthalmology 2002, 120, (3), 338-346.

Ku, H. H.; Brunk, U. T.; Sohal, R. S., Relationship between mitochondrial superoxide and hydrogen peroxide production and longevity of mammalian species. Free Radic Biol Med 1993, 15, (6), 621-7.

Kurland, J. I.; Bockman, R., Prostaglandin E production by human blood monocytes and mouse peritoneal macrophages. J. Exp. Med. 1978, 147, (3), 952-7.

Landmesser, U.; Harrison, D. G.; Drexler, H., Oxidant stress-a major cause of reduced

319 endothelial nitric oxide availability in cardiovascular disease. Eur. J. Clin. Pharmacol. 2006, 62 Suppl 13, 13-9.

LaVail, M. M., Rod outer segment disk shedding in rat retina: relationship to cyclic lighting. Science 1976, 194, 1071-1074.

Leonarduzzi, G.; Chiarpotto, E.; Biasi, F.; Poli, G., 4-Hydroxynonenal and cholesterol oxidation products in atherosclerosis. Mol Nutr Food Res 2005, 49, (11), 1044-9.

Lertsiri, S.; Shiraishi, M.; Miyazawa, T., Identification of deoxy-D-fructosyl phosphatidylethanolamine as a non-enzymic glycation product of phosphatidylethanolamine and its occurrence in human blood plasma and red blood cells. Biosci. Biotech. Biochem. 1998, 62, (5), 893-901.

Liu, J.; Itagaki, Y.; Ben-Shabat, S.; Nakanishi, K.; Sparrow, J. R., The biosynthesis of A2E, a fluorophore of aging retina, involves the formation of the precursor, A2-PE, in the photoreceptor outer segment membrane. J. Biol. Chem. 2000, 275, (38), 29354-60.

Lonn, E.; Bosch, J.; Yusuf, S.; Sheridan, P.; Pogue, J.; Arnold, J. M.; Ross, C.; Arnold, A.; Sleight, P.; Probstfield, J.; Dagenais, G. R., Effects of long-term vitamin E supplementation on cardiovascular events and cancer: a randomized controlled trial. JAMA 2005, 293, (11), 1338-47.

Lord, C.; Cook, E. H.; Leventhal, B. L.; Amaral, D. G., Autism spectrum disorders. . Neuron. 2000, 28, 355-363.

Lyczak, J. B.; Cannon, C. L.; Pier, G. B., Establishment of Pseudomonas aeruginosa infection: lessons from a versatile opportunist. Microbes Infect 2000, 2, (9), 1051-60.

Maggi, E.; Bellazzi, R.; Falaschi, F.; Frattoni, A.; Perani, G.; Finardi, G.; Gazo, A.; Nai, M.; Romanini, D.; Bellomo, G., Enhanced LDL oxidation in uremic patients: an additional mechanism for accelerated atherosclerosis? Kidney Int. 1994, 45, 876-883.

Maier, J. A.; Hla, T.; Maciag, T., Cyclooxygenase is an immediate-early gene induced by interleukin-1 in human endothelial cells. J. Biol. Chem. 1990, 265, (19), 10805-8.

Mapp, P. I.; Grootveld, M. C.; Blake, D. R., Hypoxia, oxidative stress and rheumatoid arthritis. Br Med Bull 1995, 51, (2), 419-36.

Mariella, R. P., Condensations of unsymmetrical ketones. . Condensations with ethyl formate. J. Am. Chem. Soc. 1947, 69, 2670-2672.

320

Mariella, R. P.; Godar, E., Notes - Condensations of unsymmetrical ketones. IV. Participation of methyl and methylene groups in condensation reactions. J. Org. Chem. 1957, 22, 566-568.

Marnett, L. J.; Hurd, H. K.; Hollstein, M. C.; Levin, D. E.; Esterbauer, H.; Ames, B. N., Naturally occurring carbonyl compounds are mutagens in Salmonella tester strain TA104. Mutat. Res. 1985, 148, (1-2), 25-34.

McMurray, H. F.; Parthasarathy, S.; Steinberg, D., Oxidatively modified low density lipoprotein is a chemoattractant for human T lymphocytes. J. Clin. Invest. 1993, 92, (2), 1004-8.

Meagher, E.; Rader, D. J., Antioxidant therapy and atherosclerosis: animal and human studies. Trends Cardiovasc Med 2001, 11, (3-4), 162-5.

Mendis, S.; Sobotka, P. A.; Euler, D. E., Pentane and isoprene in expired air from humans: gas-chromatographic analysis of single breath. Clin. Chem. 1994, 40, 1485-1488.

Merendino, N.; Loppi, B.; D'Aquino, M.; Molinari, R.; Pessina, G.; Romano, C.; Velotti, F., Docosahexaenoic acid induces apoptosis in the human PaCa-44 pancreatic cancer cell line by active Reduced glutathione extrusion and lipid peroxidation. Nutr Cancer 2005, 52, (2), 225-33.

Miyagi, M.; Sakaguchi, H.; Darrow, R. M.; Yan, L.; West, K. A.; Aulak, K. S.; Stuehr, D. J.; Holleyfield, J. G.; Organisciak, D. T.; Crabb, J. W., Evidence that light modulateds protein nitration in rat retina. Mol. Cell Proteomics. 2002, 1, 293-303.

Miyazaki, H.; Matsuoka, H.; Itabe, H.; Usui, M.; Ueda, S.; Okuda, S.; Imaizumi, T., Hemodialysis impairs endothelial function via oxidative stress: Effects of vitamin E coated dialyzer. Circulation 2000, 101, 1002-1006.

Money, J.; Bobrow, N. A.; Clarke, F. C., Autism and autoimmune disease: a family study. J. Autism Child Schizophrenia. 1971, 1, 146-160.

Morrow, J. D.; Hill, K. E.; Burk, R. F.; Nammour, T. M.; Badr, K. F.; Roberts, L. J., 2nd, A series of prostaglandin F2-like compounds are produced in vivo in humans by a non-cyclooxygenase, free radical-catalyzed mechanism. Proc. Natl. Acad. Sci. U. S. A. 1990, 87, (23), 9383-7.

321 Morrow, J. D.; Hill, K. E.; Burk, R. F.; Nammour, T. M.; Badr, K. F.; Roberts, L. J., A Series of Prostaglandin-F2-Like Compounds Are Produced Invivo in Humans by a Noncyclooxygenase, Free Radical-Catalyzed Mechanism. Proc. Natl. Acad. Sci. U. S. A. 1990, 87, (23), 9383-9387.

Murthi, K. K.; Friedman, L. R.; Oleinick, N. L.; Salomon, R. G., Formation of DNA-protein cross-links in mammalian cells by levuglandin E2. Biochemistry 1993, 32, (15), 4090-7.

Murthi, K. K.; Salomon, R. G.; Sternlicht, H., Levuglandin E2 inhibits mitosis and microtubule assembly. Prostaglandins 1990, 39, (6), 611-22.

Nemoto, S.; Takeda, K.; Yu, Z. X.; Ferrans, V. J.; Finkel, T., Role for mitochondrial oxidants as regulators of cellular metabolism. Mol. Cell. Biol. 2000, 20, (19), 7311-8.

Nicholls, S. J.; Shen, Z.; Fu, X.; Levison, B. S.; Hazen, S. L., Quantification of 3-nitrotyrosine levels using a benchtop ion trap mass spectrometry method. Methods Enzymol. 2005, 396, 245-266.

Nichols, F. C.; Schenkein, H. A.; Rutherford, R. B., Prostaglandin E2, prostaglandin E1 and thromboxane B2 release from human monocytes treated with C3b or bacterial lipopolysaccharide. Biochim. Biophys. Acta 1987, 927, (2), 149-57.

Nishikawa, T.; Edelstein, D.; Du, X. L.; Yamagishi, S.; Matsumura, T.; Kaneda, Y.; Yorek, M. A.; Beebe, D.; Oates, P. J.; Hammes, H. P.; Giardino, I.; Brownlee, M., Normalizing mitochondrial superoxide production blocks three pathways of hyperglycaemic damage. Nature 2000, 404, (6779), 787-90.

Nugteren, D. H., Arachidonate lipoxygenase in blood platelets. Biochim. Biophys. Acta 1975, 380, (2), 299-307.

Nugteren, D. H.; Hazelhof, E., Isolation and properties of intermediates in prostaglandin biosynthesis. Biochim. Biophys. Acta 1973, 326, (3), 448-61.

Nugteren, D. H.; Vonkeman, H.; Dorp, D. A., Non-enzymic conversion of all-cis 8, 11,

14-eicosatrienoic acid into prostaglandin E1. Rec. Trav. Chim. 1967, 86, 1237-1245.

Nugteren, D. H. V., H.; Van Dorp, D. A., Nonenzymic conversion of all-cis 8,11,14-eicosatrienoic acid into prostaglandin E1. Rec. Trav. Chim. 1967, 86, 1237-1245.

Oak, J. H.; Nakagawa, K.; Miyazawa, T., Synthetically prepared Amadori-glycated

322 phosphatidylethanolamine call trigger lipid peroxidation via free radical reactions. FEBS Lett. 2000, 481, (1), 26-30.

Odetti, P.; Fogarty, J.; Sell, D. R.; Monnier, V. M., Chromatographic quantitation of plasma and erythrocyte pentosidine in diabetic and uremic subjects. Diabetes 1992, 41, 153-159.

Odetti, P.; Robaudo, C.; Valentini, S.; Gurreri, G.; Garibaldi, S.; Angeletti, S.; Deferrari, G., Effect of a new vitamin E-coated membrane on glycoxidation during hemodialysis. Contrib Nephrol 1999, 127, 192-9.

Odetti, P.; Traverso, N.; Monacelli, F.; Menini, S.; Vazzana, J.; Tasso, B.; Pronzato, M. A.; Robaudo, C.; Deferrari, G., Vitamin E-coated filter decreases levels of free 4-hydroxyl-2-nonenal during haemodialysis sessions. Free Radic Res 2006, 40, (2), 207-12.

O'Donnell, V. B., Free Radicals and Lipid Signaling in Endothelial Cells. Antioxidants & Redox Signaling 2003, 5, 195 -203

Okura, Y.; Brink, M.; Itabe, H.; Scheidegger, K. J.; Kalangos, A.; Delafontaine, P., Circulation 2000, 102, 2680-2686.

Onorato, J. M.; Jenkins, A. J.; Thorpe, S. R.; Baynes, J. W., Pyridoxamine, an inhibitor of advanced glycation reactions, also inhibits advanced lipoxidation reactions. Mechanism of action of pyridoxamine. . J. Biol. Chem. 2000, 275, 21177-21184.

Parish, C. A.; Hashimoto, M.; Nakanishi, K.; Dillon, J.; Sparrow, J., Isolation and one-step preparation of A2E and iso-A2E, fluorophores from human retinal pigment epithelium. Proc. Natl. Acad. Sci. USA 1998, 95, (25), 14609-13.

Peng, T.; Lu, X.; Feng, Q., NADH oxidase signaling induces cyclooxygenase-2 expression during lipopolysaccharide stimulation in cardiomyocytes. FASEB J. 2005, 19, (2), 293-5.

Perez, V. L.; Yang, X.; Raybourn, M. E.; Ufret, R.; Yu, M.; Lu, L.; Salomon, R. G.; Peachey, N. S.; Hollyfield, J. G., Auto-Immune Responses to Oxidatively Altered Self Proteins in the Retina Lead to the Development of Retinal Degeneration. 2007 ARVO Annual Meeting. May 6-10, 2007, Fort Lauderdale, FL, U. S. A.

Persson, B., Brief report: A longitudinal study of quality of life and independence among adult men with autism. . J. Autism Dev. Disord 2000, 30, 61-66.

323

Peters, W.; Charo, I. F., Involvement of chemokine receptor 2 and its ligand, monocyte chemoattractant protein-1, in the development of atherosclerosis: lessons from knockout mice. Curr Opin Lipidol 2001, 12, (2), 175-80.

Poliakov, E.; Brennan, M. L.; Macpherson, J.; Zhang, R.; Sha, W.; Narine, L.; Salomon, R. G.; Hazen, S. L., Isolevuglandins, a novel class of isoprostenoid derivatives, function as integrated sensors of oxidant stress and are generated by myeloperoxidase in vivo. FASEB J. 2003, 17, (15), 2209-20.

Porter, N. A.; Weber, B. A.; Weenen, H.; Khan, J. A., Autoxidation of polyunsaturated lipids. Factors controlling the stereochemistry of product hydroperoxides J. Am. Chem. Soc. 1980, 102, 5597-5601.

Porter, N. A.; Wolf, R. A.; and Weenen, H., The free radical oxidation of polyunsaturated lecithins Lipids 1980, 15, 163-167.

Ravandi, A.; Kuksis, A.; Marai, L.; Myher, J. J., Preparation and Characterization of Glucosylated Aminoglycerophospholipids. Lipids 1995, 30, (10), 885-891.

Ravandi, A.; Kuksis, A.; Marai, L.; Myher, J. J.; Steiner, G.; Lewisa, G.; Kamido, H., Isolation and identification of glycated aminophospholipids from red cells and plasma of diabetic blood. FEBS Lett. 1996, 381, (1-2), 77-81.

Raz, A.; Wyche, A.; Needleman, P., Temporal and pharmacological division of fibroblast cyclooxygenase expression into transcriptional and translational phases. Proc. Natl. Acad. Sci. U. S. A. 1989, 86, (5), 1657-61.

Raz, A.; Wyche, A.; Siegel, N.; Needleman, P., Regulation of fibroblast cyclooxygenase synthesis by interleukin-1. J. Biol. Chem. 1988, 263, (6), 3022-8.

Requena, J. R.; Ahmed, M. U.; Fountain, C. W.; Degenhardt, T. P.; Reddy, S.; Perez, C.; Lyons, T. J.; Jenkins, A. J.; Baynes, J. W.; Thorpe, S. R., Carboxymethylethanolamine, a biomarker of phospholipid modification during the maillard reaction in vivo. J. Biol. Chem. 1997, 272, (28), 17473-17479.

Rimm, E. B.; Stampfer, M. J.; Ascherio, A.; Giovannucci, E.; Colditz, G. A.; Willett, W. C., Vitamin E consumption and the risk of coronary heart disease in men. N Engl J Med 1993, 328, (20), 1450-6.

Ristimaki, A.; Garfinkel, S.; Wessendorf, J.; Maciag, T.; Hla, T., Induction of

324 cyclooxygenase-2 by interleukin-1 alpha. Evidence for post-transcriptional regulation. J. Biol. Chem. 1994, 269, (16), 11769-75.

Roberts, L. J., 2nd; Brame, C. J.; Chen, Y.; Morrow, J. D.; Salomon, R. G., Formation of reactive products of the isoprostane pathway: isolevuglandins and cyclopentenone isoprostanes. Adv. Exp. Med. Biol. 1999, 469, 335-41.

Roberts, L. J., 2nd; Salomon, R. G.; Morrow, J. D.; Brame, C. J., New developments in the isoprostane pathway: identification of novel highly reactive gamma-ketoaldehydes (isolevuglandins) and characterization of their protein adducts. FASEB J. 1999, 13, (10), 1157-68.

Rollins, B. J.; Pober, J. S., Interleukin-4 induces the synthesis and secretion of MCP-1/JE by human endothelial cells. Am. J. Pathol. 1991, 138, (6), 1315-9.

Rollins, B. J.; Yoshimura, T.; Leonard, E. J.; Pober, J. S., Cytokine-activated human endothelial cells synthesize and secrete a monocyte chemoattractant, MCP-1/JE. Am. J. Pathol. 1990, 136, (6), 1229-33.

Roob, J. M.; Khoschsorur, G.; Tiran, A.; Horina, J. H.; Holzer, H.; Winklhofer-Roob, B., Vitamin E attenuates oxidative stress induced by intravenous iron in patients on hemodialysis. J Am Soc Nephrol 2000, 11, 539-549.

Royals, E. E.; Covington, E. R., Hydroxymethylene ketones. IV. Orientation in the condensation of methyl n-hexyl ketone with methyl formate. J. Am. Chem. Soc. 1955, 77, 3155-3157.

Rozanowska, M.; Jarvis-Evans, J.; Korytowski, W.; Boulton, M. E.; Burke, J. M.; Sarna, T., Blue light-induced reactivity of retinal age pigment. In vitro generation of oxygen-reactive species. J. Biol. Chem. 1995, 270, (32), 18825-30.

Rustici, A.; Velucchi, M.; Faggioni, R.; Sironi, M.; Ghezzi, P.; Quataert, S.; Green, B.; Porro, M., Molecular mapping and detoxification of the lipid A binding site by synthetic peptides. Science 1993, 259, (5093), 361-5.

Sakai, N.; Decatur, J.; Nakanishi, K., J. Amer. Chem. Soc. 1996, 118, 1559-1560.

Saldeen, T.; Li, D.; Mehta, J. L., Differential effects of alpha- and gamma-tocopherol on low-density lipoprotein oxidation, superoxide activity, platelet aggregation and arterial thrombogenesis. J Am Coll Cardiol 1999, 34, (4), 1208-15.

325 Salomon, M. F.; Salomon, R. G., 2,3-Dioxabicyclo[2.2.1]heptane. The strained cyclic peroxide nucleus of prostaglandin endoperoxides. J. Am. Chem. Soc. 1977, 99, 3501.

Salomon, R.; Kaur, K.; Batyreva, E., Isolevuglandin-protein adducts in oxidized low density lipoprotein and human plasma: a strong connection with cardiovascular disease. Trends Cardiovasc Med 2000, 10, (2), 53-9.

Salomon, R.; Kaur, K.; Podrez, E.; Hoff, H.; Krushinsky, A.; Sayre, L., HNE-derived 2-pentylpyrroles are generated during oxidation of LDL, are more prevalent in blood plasma from patients with renal disease or atherosclerosis, and are present in atherosclerotic plaques. Chem. Res. Toxicol. 2000, 13, (7), 557-64.

Salomon, R. G., Distinguishing levuglandins produced through the cyclooxygenase and isoprostane pathways. Chem. Phys. Lipids 2005, 134, (1), 1-20.

Salomon, R. G., Levuglandins and isolevuglandins: stealthy toxins of oxidative injury. Antioxid Redox Signal 2005, 7, (1-2), 185-201.

Salomon, R. G.; Batyreva, E.; Kaur, K.; Sprecher, D. L.; Schreiber, M. J.; Crabb, J. W.; Penn, M. S.; DiCorletoe, A. M.; Hazen, S. L.; Podrez, E. A., Isolevuglandin-protein adducts in humans: products of free radical-induced lipid oxidation through the isoprostane pathway. Biochim. Biophys. Acta 2000, 1485, (2-3), 225-35.

Salomon, R. G.; Jirousek, M. R.; Ghosh, S.; Sharma, R. B., Prostaglandin endoperoxides 21. Covalent binding of levuglandin E2 with proteins. Prostaglandins 1987, 34, (5), 643-56.

Salomon, R. G.; Kaur, K.; Batyreva, E., Isolevuglandin-protein adducts in oxidized low density lipoprotein and human plasma: A strong connection with cardiovascular disease. Trends in Cardiovascular Medicine 2000, 10, (2), 53-59.

Salomon, R. G.; Kaur, K.; Podrez, E.; Hoff, H. F.; Krushinsky, A. V.; Sayre, L. M., HNE-derived 2-pentylpyrroles are generated during oxidation of LDL, are more prevalent in blood plasma from patients with renal disease or atherosclerosis, and are present in atherosclerotic plaques. Chem. Res. Toxicol. 2000, 13, (7), 557-64.

Salomon, R. G.; Miller, D. B.; Zagorski, M. G.; and Coughlin, D. J., Solvent induced

fragmentation of prostaglandin endoperoxides. New aldehyde products from PGH2 and a novel intramolecular 1,2-hydride shift during endoperoxide fragmentation in aqueous solution. J. Am. Chem. Soc. 1984, 106, 6049-6060.

326 Salomon, R. G.; Miller, D. B.; Zagorski, M. G.; Coughlin, D. J., Solvent induced

fragmentation of prostaglandin endoperoxides. New aldehyde products from PGH2 and a novel intramolecular 1,2-hydride shift during endoperoxide fragmentation in aqueous solution. J. Am. Chem. Soc. 1984, 106, 6049-6060.

Salomon, R. G.; Sha, W.; Brame, C.; Kaur, K.; Subbanagounder, G.; O'Neil, J.; Hoff, H. F.; Roberts, L. J., 2nd, Protein adducts of iso[4]levuglandin E2, a product of the isoprostane pathway, in oxidized low density lipoprotein. J. Biol. Chem. 1999, 274, (29), 20271-80.

Salomon, R. G.; Sha, W.; Brame, C.; Kaur, K.; Subbanagounder, G.; O'Neil, J.; Hoff, H. F.; Roberts, L. J., Protein adducts of iso[4]levuglandin E-2, a product of the isoprostane pathway, in oxidized low density lipoprotein. J. Biol. Chem. 1999, 274, (29), 20271-20280.

Salomon, R. G.; Subbanagounder, G.; O’neil, J.; Kaur, K.; Smith, M. A.; Hoff, H. F.; Perry, G.; and Monnier, V. M., Chem. Res. Toxicol. 1997, 10, 536-545.

Salomon, R. G.; Subbanagounder, G.; O'Neil, J.; Kaur, K.; Smith, M. A.; Hoff, H. F.; Perry, G.; Monnier, V. M., Levuglandin E2-protein adducts in human plasma and vasculature. Chem. Res. Toxicol. 1997, 10, (5), 536-45.

Salomon, R. G.; Subbanagounder, G.; Singh, U.; O'Neil, J.; and Hoff, H., Oxidation of LDL produces levuglandin-protein adducts. Chem. Res. Toxicol. 1997, 10, 750-759.

Salomon, R. G.; Subbanagounder, G.; Singh, U.; O'Neil, J.; Hoff, H. F., Oxidation of low-density lipoproteins produces levuglandin-protein adducts. Chem. Res. Toxicol. 1997, 10, (7), 750-9.

Salomon, R. G. M., D. B.; Zagorski, M. G.; and Coughlin, D.J., Prostaglandin Endoperoxides. 14. Solvent-induced fragmentation of prostaglandin endoperoxides. New aldehyde products from PGH2 and a novel intramolecular 1,2-hydride shift during endoperoxide fragmentation in aqueous solution. J. Am. Chem. Soc. 1984, 106, 6049-6060.

Sambrano, G. R.; Parthasarathy, S.; Steinberg, D., Recognition of oxidatively damaged erythrocytes by a macrophage receptor with specificity for oxidized low density lipoprotein. Proc. Natl. Acad. Sci. U. S. A. 1994, 91, (8), 3265-9.

Samsel, E. G.; Kochi, J. K., Oxidative alkylation of cobalt complexes with hydrazines. Inorg. Chem. 1986, 25, 2450-2457.

327

Satoh, M.; Yamasaki, Y.; Nagake, Y.; Kasahara, J.; Hashimoto, M.; Nakanishi, N.; Makino, H., Oxidative stress is reduced by the long-term use of vitamin E-coated dialysis filters. Kidney Int. 2001, 59, (5), 1943-50.

Sayre, L. M.; Arora, P. K.; Iyer, R. S.; Salomon, R. G., Pyrrole formation from 4-hydroxynonenal and primary amines. Chem. Res. Toxicol. 1993, 6, (1), 19-22.

Sayre, L. M.; Sha, W.; Xu, G.; Kaur, K.; Nadkarni, D.; Subbanagounder, G.; Salomon, R. G., Immunochemical evidence supporting 2-pentylpyrrole formation on proteins exposed to 4-hydroxy-2-nonenal. Chem. Res. Toxicol. 1996, 9, (7), 1194-201.

Sayre, L. M.; Sha, W.; Xu, G. Z.; Kaur, K.; Nadkarni, D.; Subbanagounder, G.; Salomon, R. G., Immunochemical evidence supporting 2-pentylpyrrole formation on proteins exposed to 4-hydroxy-2-nonenal. Chem. Res. Toxicol. 1996, 9, (7), 1194-1201.

Schauenstein, E.; Esterbauer, H., Formation and properties of reactive aldehydes. Ciba Found Symp 1978, (67), 225-44.

Schauenstein, E.; Esterbauer, H.; & Zollner, H., Aldehydes in Biological Systems, 1977, Pion, London.

Schultz, C. L.; Morck, D. W.; McKay, S. G.; Olson, M. E.; Buret, A., Lipopolysaccharide induced acute red eye and corneal ulcers. Exp. Eye Res. 1997, 64, (1), 3-9.

Schutt, F.; Bergmann, M.; Holz, F. G.; Dithmar, S.; Volcker, H. E.; Kopitz, J., Accumulation of A2-E in mitochondrial membranes of cultured RPE cells. Graefes. Arch. Clin. Exp. Ophthalmol. 2006.

Schwing, W.; Erhard, P.; Hollamon, C.; Weigel, K.; Blankschaen, S.; Anderson, J.; Siegal, C.; Seaman, D.; Valente, J.; DeOreo, P.; Weiss, M., Thrombotic events and markers of oxidation and inflammation in hemodialysis. Hemodialysis Int 2004, 8, 338-343.

Sell, D. R., and Monnier, V. M., Structural elucidation of a fluorescent cross link from human senescent extracellular matrix: implication of pentoses in the aging process. . J. Biol. Chem. 1989, 264, 21597-21602.

Sheppeck, J. E.; Kar, H.; Hong, H., A convenient and scaleable procedure for removing the Fmoc group in solution. Tetrahedron Lett. 2000, 41, (28), 5329-5333.

328 Siems, W.; Grune, T., Intracellular metabolism of 4-hydroxynonenal. Mol Aspects Med 2003, 24, (4-5), 167-75.

Siems, W.; Quast, S.; Peter, D.; Augustin, W.; Carluccio, F.; Grune, T.; Sevanian, A.; Hampl, H.; Wiswedel, I., Oxysterols are increased in plasma of end-stage renal disease patients. Kidney Blood Press Res 2005, 28, (5-6), 302-6.

Simons, L. A.; von Konigsmark, M.; Simons, J.; Stocker, R.; Celermajer, D. S., Vitamin E ingestion does not improve arterial endothelial dysfunction in older adults. Atherosclerosis 1999, 143, (1), 193-9.

Singh, S. P.; Kumar, D.; Kumar, D., Conformation and ortho steric effects in a series of 2-(pyrazol-1-yl) quinolines. J. Heterocyclic Chem. 1996, 33, 323-326.

Siqueira, A. F.; Abdalla, D. S.; Ferreira, S. R., [LDL: from metabolic syndrome to instability of the atherosclerotic plaque]. Arq Bras Endocrinol Metabol 2006, 50, (2), 334-43.

Skinner, E. R.; Watt, C.; Besson, J. A.; Best, P. V., Differences in the fatty acid composition of the grey and white matter of different regions of the brains of patients with Alzheimer's disease and control subjects. Brain 1993, 116 ( Pt 3), 717-25.

Smith, E. B.; Jensen, H. B., Autoxidation of three 1-alkylpyrroles. J. Org. Chem. 1967, 32, 3330-3334.

Smith, K. S.; Lee, C. L.; Ridlington, J. W.; Leonard, S. W.; Devaraj, S.; Traber, M. G., Vitamin E supplementation increases circulating vitamin E metabolites tenfold in end-stage renal disease patients. Lipids 2003, 38, (8), 813-9.

Smith, P. K.; Krohn, R. I.; Hermanson, G. T.; Mallia, A. K.; Gartner, F. H.; Provenzano, M. D.; Fujimoto, E. K.; Goeke, N. M.; Olson, B. J.; Klenk, D. C., Measurement of Protein Using Bicinchoninic Acid Anal. Chem. 1985, 150, 76-85.

Sohal, R. S.; Agarwal, S.; Dubey, A.; Orr, W. C., Protein oxidative damage is associated with life expectancy of houseflies. Proc. Natl. Acad. Sci. USA 1993, 90, 7255-7259.

Sohal, R. S.; Ku, H. H.; Agarwal, S., Biochemical correlates of longevity in two closely related rodent species. Biochem. Biophys. Res. Commun. 1993, 196, (1), 7-11.

Sohal, R. S.; Sohal, B. H.; Orr, W. C., Mitochondrial superoxide and hydrogen peroxide

329 generation, protein oxidative damage, and longevity in different species of flies. Free Radic Biol Med 1995, 19, (4), 499-504.

Sommerburg, O.; Zang, L.-Y.; van Kuijk, F., Simultaneous detection of carotenoids and vitamin E in human plasma. J Chromatogr B 1997, 695, 209-215.

Sparrow, J. R.; Cai, B., Blue light-induced apoptosis of A2E-containing RPE: involvement of caspase-3 and protection by Bcl-2. Invest. Ophthalmol. Vis. Sci. 2001, 42, (6), 1356-62.

Sparrow, J. R.; Nakanishi, K.; Parish, C. A., The lipofuscin fluorophore A2E mediates blue light-induced damage to retinal pigmented epithelial cells. Invest. Ophthalmol. Vis. Sci. 2000, 41, (7), 1981-9.

Sparrow, J. R.; Parish, C. A.; Hashimoto, M.; Nakanishi, K., A2E, a lipofuscin fluorophore, in human retinal pigmented epithelial cells in culture. Invest. Ophthalmol. Vis. Sci. 1999, 40, (12), 2988-2995.

Sparrow, J. R.; Zhou, J.; Ben-Shabat, S.; Vollmer, H.; Itagaki, Y.; Nakanishi, K., Involvement of oxidative mechanisms in blue-light-induced damage to A2E-laden RPE. Invest. Ophthalmol. Vis. Sci. 2002, 43, (4), 1222-7.

Spiteller, G., Chem. Phys. Lipids 1998, 95, 105-62.

Spiteller, G., The relation of lipid peroxidation processes with atherogenesis: a new theory on atherogenesis. Mol Nutr Food Res 2005, 49, (11), 999-1013.

Stadtman, E. R., Protein oxidation and aging. Science 1992, 257, 1220-1224.

Stadtman, E. R.; Oliver, C. N., Metal-catalyzed oxidation of proteins. Physiological consequences. J. Biol. Chem. 1991, 266, 2005-2008.

Stampfer, M. J.; Hennekens, C. H.; Manson, J. E.; Colditz, G. A.; Rosner, B.; Willett, W. C., Vitamin E consumption and the risk of coronary disease in women. N Engl J Med 1993, 328, (20), 1444-9.

Steinberg, D., J. Biol. Chem. 1997, 272, 20963-20966.

Steinberg, D.; Parthasarathy, S.; Carew, T. E.; Khoo, J. C.; Witztum, J. L., Beyond cholesterol. Modifications of low-density lipoprotein that increase its atherogenicity. N Engl J Med 1989, 320, (14), 915-24.

330

Stephens, N.; Parsons, A.; Shofield, P.; Kelly, F.; Cheeseman, K.; Mitchinson, M.; Brown, M., Randomised controlled trial of vitamin E in patients with coronary disease: Cambridge Heart Antioxidant Study (CHAOS). Lancet 1996, 347, 781-786.

Stillway, L. W.; Harmon, S. J., A procedure for detecting phosphonolipids on thin-layer chromatograms. J. Lipid Res. 1980, 21, (8), 1141-3.

Stitt, A.; Gardiner, T. A.; Alderson, N. L.; Canning, P. F., N.; ; Duffy, N.; Boyle, C.; Januszewski, A. S.; Chachich, M.; Baynes, J. W.; Thorpe, S. R., The AGE inhibitor pyridoxamine inhibits development of retinopathy in experimental diabetes. Diabetes 2002, 51, 2826-2832.

Stone, E. M.; Sheffield, V. C.; Hageman, G. S., Molecular genetics of age-related macular degeneration. Human Molecular Genetics 2001, 10, (20), 2285-2292.

Subbanagounder, G.; Deng, Y.; Borromeo, C.; Dooley, A. N.; Berliner, J. A.; Salomon, R. G., Hydroxy alkenal phospholipids regulate inflammatory functions of endothelial cells. Vascul Pharmacol 2002, 38, (4), 201-9.

Sun, H.; Nathans, J., ABCR, the ATP-binding cassette transporter responsible for Stargardt macular dystrophy, is an efficient target of all-trans-retinal-mediated photooxidative damage in vitro. Implications for retinal disease. J. Biol. Chem. 2001, 276, (15), 11766-74.

Sweeten, T. L.; Bowyer, S. L.; Posey, D. J.; Halberstadt, G. M.; McDougle, C. J., Increased prevalence of familial autoimmunity in probands with pervasive developmental disorders. Pediatrics. 2003, 112, e420.

Szweda, L. I.; Uchida, K.; Tsai, L.; Stadtman, E. R., Inactivation of glucose-6-phosphate dehydrogenase by 4-hydroxy-2-nonenal. Selective modification of an active-site lysine. J. Biol. Chem. 1993, 268, (5), 3342-7.

Tappel, A. L., Lipid peroxidation and fluorescent molecular damage to membranes. Pathobiology of Cell Membranes, edited by B. F. Trump and A. U. Arstila. 1975, p 145-170.

Tappel, A. L., Measurement of and protection from in vivo lipid peroxidation. In: Pryor WA, ed,. Free Radicals in Biology 1980, vol 4. New York: Academic Press, 2-47.

Tenchov, B.; Vescio, E. M.; Sprott, G. D.; Zeidel, M. L.; Mathai, J. C., Salt tolerance of

331 archaeal extremely halophilic lipid membranes. J. Biol. Chem. 2006, 281, (15), 10016-23.

Toshniwal, P. K.; Zarling, E. J., Evidence for increased lipid peroxidation in multiple sclerosis. Neurochem. Res. 1992, 17, (2), 205-7.

Traber, M. G.; Arai, H., Molecular mechanisms of vitamin E transport. Annu Rev Nutr 1999, 19, 343-355.

Tsuji, K.; Kawai, Y.; Kato, Y.; Osawa, T., Formation of N-(hexanoyl)ethanolamine, a novel phosphatidylethanolamine adduct, during the oxidation of erythrocyte membrane and low-density lipoprotein. Biochem. Biophys. Res. Commun. 2003, 306, (3), 706-711.

Uchida, K., Amino Acids 2003, 25, 249-257.

Uchida, K.; Szweda, L. I.; Chae, H. Z.; Stadtman, E. R., Immunochemical detection of 4-hydroxynonenal protein adducts in oxidized hepatocytes. Proc. Natl. Acad. Sci. U. S. A. 1993, 90, (18), 8742-6.

Uebel, R.; Jaarsveld, P. v.; Hawtrey, A. O., Preparation of biotinylated spermine and the improved synthesis of some related amino derivatives. Med. Sci. Res. 1990, 18, 777-778. van der Schaft, T. L.; de Bruijn, W. C.; Mooy, C. M.; Ketelaars, D. A.; de Jong, P. T., Element analysis of the early stages of age-related macular degeneration. Arch. Ophthalmol. 1992, 110, (3), 389-94.

Vojdani, A.; Campbell, A. W.; Anyanwu, E.; Kashanian, A.; Bock, K.; Vojdani, E., Antibodies to neuron-specific antigens in children with autism: possible cross-reaction with encephalitogenic proteins from milk, Chlamydia pneumoniae and Streptococcus group A. J. Neuroimmunol. 2002, 129, (1-2), 168-177.

Wakefield, A. J.; Murch, S. H.; Anthony, A., et al. , Ileal-lymphoid-nodular hyperplasia, nonspecific colitis, and pervasive developmental disorder in children. . Lancet. 1998, 351, 637-641.

Walsh, W. J.; Usman, A.; Tarpey, J., American Psychiatric Association Annual Meeting; New Orleans, LA. 2001.

Wang, N.; Anderson, R. E., Enrichment of polyunsaturated fatty acids from rat retinal pigment epithelium to rod outer segments. Curr Eye Res 1992, 11, (8), 783-91.

Wanner, C.; Bahner, U.; Mattern, R.; Lang, D.; Passlick-Deetjen, J., Effect of dialysis

332 flux and membrane material on dyslipidaemia and inflammation in haemodialysis patients. Nephrol Dial Transplant 2004, 19, (10), 2570-5.

Waring, R. H.; Klovrza, L. V., Sulphur Metabolism in Autism. J. Nutri. Enviro. Med. 2000, 10, 25-32.

Warren, R. P.; Foster, A.; Margaretten, N. C., Immune abnormalities in patients with autism. J. Autism Dev. Disord. 1987, 16, 189-197.

Wechter, W. J.; Kantoci, D.; Murray, E. D., Jr.; D'Amico, D. C.; Jung, M. E.; Wang, W. H., A new endogenous natriuretic factor: LLU-alpha. Proc. Natl. Acad. Sci. U. S. A. 1996, 93, (12), 6002-7.

Weiss, M.; Erhard, P.; Kader-Attia, F.; Wu, Y.; DeOreo, P.; Araki, A.; Monnier, V., Mechanisms for the formation of advanced glycation end products (AGEs) in end stage renal disease (ESRD). Kidney Int. 2000, 57, 2571-2585.

West, K. A.; Yan, L.; Miyagi, M.; Crabb, J. S.; Marmorstein, A. D.; Marmorstein, L.; Crabb, J. W., Proteome survey of proliferating and differentiating rat RPE-J cells. Exp. Eye Res. 2001, 73, (4), 479-91.

Wielkoszynski, T.; Gawron, K.; Strzelczyk, J.; Bodzek, P.; Zalewska-Ziob, M.; Trapp, G.; Srebniak, M.; Wiczkowski, A., Cellular toxicity of oxycholesterols. BioEssays 2006, 28, (4), 387-398.

Williams, R. A.; Brody, B. L.; Thomas, R. G.; Kaplan, R. M.; Brown, S. I., The psychosocial impact of macular degeneration. Arch Ophthalmol 1998, 116, (4), 514-20.

Winklhofer-Roob, B. M.; Meinitzer, A.; Maritschnegg, M.; Roob, J. M.; Khoschsorur, G.; Ribalta, J.; Sundl, I.; Wuga, S.; Wonisch, W.; Tiran, B.; Rock, E., Effects of vitamin E depletion/repletion on biomarkers of oxidative stress in healthy aging. Ann. N. Y. Acad. Sci. 2004, 1031, 361-4.

Winklhofer-Roob, B. M.; van't Hof, M. A.; Shmerling, D. H., Reference values for plasma concentrations of vitamin E and A and carotenoids in a Swiss population from infancy to adulthood, adjusted for seasonal influences. Clin Chem 1997, 43, (1), 146-53.

Wiswedel, I.; Hirsch, D.; Carluccio, F.; Hampl, H.; Siems, W., F2-isoprostanes as biomarkers of lipid peroxidation in patients with chronic renal failure. Biofactors 2005, 24, (1-4), 201-8.

333 Witko-Sarsat, V.; Friedlander, M.; Capeillere-Blandin, C.; Nguyen-Khoa, T.; Nguyen, A.; Zingraff, J.; Jungers, P.; Descamps-Latscha, B., Advanced oxidative protein products as a novel marker of oxidative stress in uremia. Kidney Int. 1996, 49, 1304-1313.

Witting, P. K.; Upston, J. M.; Stocker, R., The molecular action of alpha-tocopherol in lipoprotein lipid peroxidation. Pro- and antioxidant activity of vitamin E in complex heterogeneous lipid emulsions. Subcell Biochem 1998, 30, 345-90.

Witz, G., Biological interactions of alpha,beta-unsaturated aldehydes. Free Radic. Biol. Med. 1989, 7, (3), 333-49.

Xu, G.; Sayre, L. M., Chem. Res. Toxicol. 1998, 11, 247-251.

Xu, G.; Sayre, L. M., Structural elucidation of a 2:2 4-ketoaldehyde-amine adduct as a model for lysine-directed cross-linking of proteins by 4-ketoaldehydes. Chem. Res. Toxicol. 1999, 12, (9), 862-8.

Yamauchi, R.; Ozaki, K.; Shimoyamada, M.; Kato, K., Iron-catalyzed reaction products of alpha-tocopherol with 1-palmitoyl-2-linoleoyl-3-sn-phosphatidylcholine (13S)-hydroperoxide. Chem. Phys. Lipids 2002, 114, (2), 193-201.

Yamauchi, R.; Yagi, Y.; Kato, K., Oxidation of alpha-tocopherol during the peroxidation of dilinoleoylphosphatidylcholine in liposomes. Biosci Biotechnol Biochem 1996, 60, (4), 616-20.

Yla-Herttuala, S.; Palinski, W.; Rosenfeld, M. E.; Parthasarathy, S.; Carew, T. E.; Butler, S.; Witztum, J. L.; Steinberg, D., Evidence for the presence of oxidatively modified low density lipoprotein in atherosclerotic lesions of rabbit and man. J. Clin. Invest. 1989, 84, (4), 1086-95.

Young, R. W., The renewal of photoreceptor cell outer segments. J. Cell. Biol. 1967, 33, 61-72.

Young, R. W.; and Bok, D., Participation of the retinal pigment epithelium in the rod outer segment renewal process. J. Cell. Biol. 1969, 42, 392-403.

Zagol-Ikapitte, I.; Masterson, T. S.; Amarnath, V.; Montine, T. J.; Andreasson, K. I.; Boutaud, O.; Oates, J. A., Prostaglandin H(2)-derived adducts of proteins correlate with Alzheimer's disease severity. J. Neurochem. 2005, 94, (4), 1140-5.

Zamora, R.; Hidalgo, F. J., Phosphatidylethanolamine modification by oxidative stress

334 product 4, 5 (E)-epoxy-2 (E)-heptenal. Chem. Res. Toxicol. 2003, 16, (12), 1632-1641.

Zarkovic, K., 4-hydroxynonenal and neurodegenerative diseases. Mol Aspects Med 2003, 24, (4-5), 293-303.

Zarkovic, N., 4-hydroxynonenal as a bioactive marker of pathophysiological processes. Mol Aspects Med 2003, 24, (4-5), 281-91.

ZARLING, E. J.; Mobarhan, S.; Bowen, P.; Kamath, S., Pulmonary pentane excretion increases with age in healthy subjects. Mech. Ageing Dev. 1993, 67, 141-147.

Zhang, R.; Brennan, M. L.; Shen, Z.; MacPherson, J. C.; Schmitt, D.; Molenda, C. E.; Hazen, S. L., Myeloperoxidase functions as a major enzymatic catalyst for initiation of lipid peroxidation at sites of inflammation. J. Biol. Chem. 2002, 277, (48), 46116-22.

Zhang, Y.; Ramos, B. F.; Jakschik, B. A., Neutrophil recruitment by tumor necrosis factor from mast cells in immune complex peritonitis. Science 1992, 258, (5090), 1957-9.

Zhou, J.; Cai, B.; Jang, Y. P.; Pachydaki, S.; Schmidt, A. M.; Sparrow, J. R., Mechanisms for the induction of HNE- MDA- and AGE-adducts, RAGE and VEGF in retinal pigment epithelial cells. Exp. Eye Res. 2005, 80, (4), 567-80.

Zhu, M.; Spink, D. C.; Yan, B.; Bank, S.; DeCaprio, A. P., Formation and structure of cross-linking and monomeric pyrrole autoxidation products in 2,5-hexanedione-treated amino acids, peptides, and protein. Chem. Res. Toxicol. 1994, 7, (4), 551-8.

Zieseniss, S.; Zahler, S.; Muller, I.; Hermetter, A.; Engelmann, B., Modified phosphatidylethanolamine as the active component of oxidized low density lipoprotein promoting platelet prothrombinase activity. J. Biol. Chem. 2001, 276, (23), 19828-19835. http://www.iom.edu/CMS/3793/4705/20155.aspx.

Bio-rad Protein Assay Instruction Manual. www.bio-rad.com/LifeScience/pdf/Bulletin_9005.pdf

Free radical introduction. http://www.exrx.net/Nutrition/Antioxidants/Introduction.html.

Instructions of modified Lowry protein assay kit. www.piercenet.com.

Introduction to lipid peroxidation. http://www.cyberlipid.org/perox/oxid0002.htm.

335 Dietary supplementation with n-3 polyunsaturated fatty acids and vitamin E after myocardial infarction: results of the GISSI-Prevenzione trial. Gruppo Italiano per lo Studio della Sopravvivenza nell'Infarto miocardico. Lancet 1999, 354, (9177), 447-55.

MRC/BHF Heart Protection Study of antioxidant vitamin supplementation in 20,536 high-risk individuals: a randomised placebo-controlled trial. Lancet 2002, 360, (9326), 23-33.

336