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Henoch-Schönlein Purpura Associated with Solid-Organ Malignancies: Three Case Reports and a Literature Review

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Henoch-Schönlein Purpura Associated with Solid-Organ Malignancies: Three Case Reports and a Literature Review Included in the theme issue: INFLAMMATORY SKIN DISEASES Acta Derm Venereol 2012; 92: 388–392 Acta Derm Venereol 2012; 92: 339–409 CLINICAL REPORT Henoch-Schönlein Purpura Associated With Solid-organ Malignancies: Three Case Reports and a Literature Review Joshua O. PODJASEK, David A. WETTER, Mark R. PITTELKOW and David A. WADA Department of Dermatology, Mayo Clinic, Rochester, MN, USA Adult Henoch-Schönlein purpura (HSP) is rarely asso- Table I. Summary of characteristics of 47 previously reported ciated with solid-organ malignancies. We describe here patients with Henoch-Schönlein purpura associated with solid- a three adult patients with HSP diagnosed within 3 months organ malignancy of the diagnosis of associated solid-organ malignancies, Characteristic Value including pulmonary, prostate, and renal carcinomas. Age, years, mean 62b Two patients had complete remission with a combina- Men, n (%) 33 (70) c tion of immunosuppressive therapies and treatment of Type of solid-organ malignancy (n = 53) , n (%) Lung 14 (26) the associated malignancy. The third patient had partial Prostate 6 (11) remission with immunosuppressive therapies, but never Kidney 5 (9) received treatment for the associated malignancy and Gastric 4 (8) did not achieve complete remission before his death 10 Breast 3 (6) Thyroid 3 (6) months after diagnosis of HSP. These cases suggest that Carcinoid 2 (4) HSP associated with solid-organ malignancies may be Maxillary 2 (4) resistant to immunosuppressive therapies without treat- Cervical 2 (4) ment of the associated malignancy. Therefore, evaluation Colon 2 (4) Epiglottic, Hypopharyngeal 2 (4) for solid-organ malignancies should be considered in Esophageal 1 (2) adult patients without an identifiable cause of HSP, espe- Anal, Rectal 2 (4) cially if the disease is not self-limited or does not respond Ovarian, Endometrial 2 (4) appropriately to treatment. Key words: coagulation; cu- Hepatocellular, Cholangiocarcinoma 2 (4) Schwannoma 1 (2) taneous disease; immunopathology; immunofluorescence; Onset of cutaneous vasculitis in relation to microvascular occlusion syndrome; purpura; vasculitis. malignancy (n = 53)c, n (%) Before 14 (26) (Accepted October 3, 2011.) Synchronousd 19 (36) After 20 (38) Acta Derm Venereol 2012; 92: 388–392. Response of cutaneous vasculitis to treatment, n (%) Remission with immunosuppressive agents 3 (6) Remission with treatment of cancer 9 (19) David A. Wetter, Department of Dermatology, Mayo Remission with combined treatment 4 (9) Clinic, 200 First St SW, Rochester, MN 55905, USA. No remission 0 E-mail: [email protected] Unknown 31 (66) Outcome, n (%) Alive 8 (17) Deceased 14 (30) We recently described 17 cases of cutaneous small vessel Unknown 25 (53) vasculitis (CSVV) associated with solid-organ malignan- aThree cases described by Fain et al. (25) were not included because indivi- cies (1). In that study, CSVV was equivalent to leukocy- dual patient information was not available. toclastic vasculitis or hypersensitivity vasculitis (1), per bTwo cases had no record of patient age or sex. the inclusion criteria of Fiorentino (2). Similar to CSVV, cSix patients (cases 14, 18, 27, 33, 34, and 47 in Table SI) had 2 separate malignancies associated with HSP. Henoch-Schönlein purpura (HSP) has also previously d been associated with malignancy. However, in contrast to Occurred within 1 month of each other. CSVV, HSP has been reported to occur more commonly with solid-organ malignancies than with hematological HSP associated with solid-organ malignancies seen at malignancies (3, 4). In particular, pulmonary, prostatic, Mayo Clinic over the previous 13 years. and renal carcinomas have been most commonly as- sociated with the development of HSP (4). In total, 47 CASE REPORTS cases of HSP related to solid-organ malignancy have been This study was approved by the Mayo Clinic Institutional reported previously (Table I and Table SI; available from: Review Board. http://www.medicaljournals.se/acta/content/?doi=10.234 Case 1. A 47-year-old man presented with palpable purpura of 0/00015555-1288). We report here 3 additional cases of one week’s duration involving his lower extremities (Fig. 1A, Acta Derm Venereol 92 © 2012 The Authors. doi: 10.2340/00015555-1288 Journal Compilation © 2012 Acta Dermato-Venereologica. ISSN 0001-5555 lower lower extremity lesion indicated leukocytoclastic vasculitis, and hematuria and proteinuria on urinalysis. Skin biopsy of a ≤ (normal mm/h 56 of rate sedimentation erythrocyte an included purpura the of evaluation during values laboratory positive Pertinent metastases. nodal for cisplatin and etoposide with chemotherapy and radiotherapy subsequent and tumor the carcinoma had included bronchoscopy with laser ablation of the of Treatment T4N2M0). IIIb, (stage lung the of carcinoma she had received a diagnosis of months squamous earlier, cell purpura on her bilateral lower extremities (Table II). Three Case 2. has shownnosignsofrecurrenceafter28monthsfollow-up. HSP the and later, months 2 tapered was dapsone oral The loped. deve lesions new No improve. to continued symptoms systemic nephropathy. and C3,consistentwithIgA analysis of the glomeruli showed mesangial staining for IgA the initial diagnosis of Immunofluorescent HSP. histological after weeks 5 approximately nephrectomy partial underwent He revealed clear cell renal cell carcinoma (stage I, T1aNXM0). evaluation Further identified. was mass renal enhancing solid, domen to evaluate his abdominal pain and diarrhea. A 2-cm, continued toimprovethroughouta4-weekprednisonetaper. symptoms and lesions skin The prednisone. oral starting of days 2 within swelling uvular and arthritis, pain, abdominal rash, his in improvement significant had he initiated; was daily orally mg probably associated Treatment with with prednisone HSP. 60 evaluation determined that he had Otolaryngological uvular oropharynx. his swelling in fullness that of sensation was a and irritation experienced he addition, In diarrhea. and nausea, pain, abdominal with along hands, and ankles, knees, his in developed pain arthritic Concomitantly, 1B). (Fig. extremities lower distal number of large, crusted, necrotic increasing An lesions extremities. developed upper bilateral on and trunk the his involve fluocinonide 0.05%creamandtacrolimus0.1%ointment. topical as well as daily, twice orally mg 25 dapsone with begun was treatment and made, was HSP probable of diagnosis A turia. microhema revealed Urinalysis vessels. blood dermal superficial on samples of lesional skin revealed deposition of IgA within cytoclastic vasculitis. Direct immunofluorescence performed leuko demonstrated lesions representative of Biopsies II). Table plaques withsurroundingviolaceouserythemaalongtheanteriortibia. necrotic coalescent, occasionally Clustered, B. extremities. lower bilateral the 1. Fig. After the partial nephrectomy, the patient’s skin lesions and The patient underwent computed tomography of the ab Throughout the next several weeks, the lesions spread to A. Palpable purpura and several larger, necrotic, purpuric plaques on plaques purpuric necrotic, larger, several and purpura Palpable A. A 71-year-old woman was evaluated for palpable 22 mm/h) mm/h) 22 - - - - Table II. Characteristics of patients with Henoch-Schönlein purpura (HSP) associated with solid-organ malignancy Type of Age, Presenting features Onset of vasculitis in solid-organ Abnormal laboratory Treatment of solid- Response to Outcome, Case years/sex of HSP relation to malignancy malignancy valuesa Treatment of vasculitis organ malignancy treatment follow-upb Comment 1c 47/M Purpura, arthritis, Synchronousd Renal Mildly increased Prednisone, dapsone, Surgery Remissione Alive, Uvular swelling believed abdominal pain, carcinoma RF level (negative fluocinolone 0.05% 28 months to be secondary to HSP nausea, diarrhea CCP antibodies), cream, tacrolimus 0.1% per otolaryngologist microhematuria ointment evaluation malignancy andsolid-organ HSP 2 71/F Purpura, arthritis 3 months after Lung Increased ESR, Prednisone Surgery, Remissione Deceased, None carcinoma microhematuria, chemotherapy, 14 months proteinuria irradiation 3 68/M Purpura, bilateral Synchronousd Prostate Increased ESR Prednisone, HCQ, None Partial Deceased, Bilateral pulmonary pulmonary carcinoma and CRP levels, triamcinolone 0.1% remissionf 10 months infiltrates believed to be infiltrates, arthritis decreased C3 level, cream secondary to HSP per microhematuria pulmonologist evaluation aSkin biopsy findings showed leukocytoclastic vasculitis in all cases. Acta Derm Venereol 92 Acta DermVenereol bFrom diagnosis of HSP. cBriefly reported in Podjasek et al. (1). dOccurred within 1 month of each other. eAfter combination of cancer treatment and immunosuppressive therapy. fAfter treatment of vasculitis with glucocorticoids or other immunosuppressive agent. CCP: cyclic citrullinated peptide; CRP: C-reactive protein; DIF: direct immunofluorescence; ESR: erythrocyte sedimentation rate; HCQ: hydroxychloroquine; RF: rheumatoid factor. 389 390 J. O. Podjasek et al. and direct immunofluorescence demonstrated IgA and C3 HSP is 87.1% sensitive and 87.7% specific when at deposition within papillary dermal blood vessels. Moreover, least 2 of the following features are present: palpable on renal biopsy to evaluate the hematuria and proteinuria, im- munofluorescent histological analysis showed segmental and purpura, bowel angina, age 20 years or younger at diffuse granular mesangial staining for IgA and C3, consistent onset, and histological changes of leukocytoclastic
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