DOCSLIB.ORG

Autoimmune Retinopathy: a Review Aristófanes Mendonça Canamary Jr.1* , Walter Yukihiko Takahashi2 and Juliana Maria Ferraz Sallum1

Total Page：16

File Type：pdf, Size：1020Kb

Download full-text PDF Read full-text
Autoimmune Retinopathy: a Review Aristófanes Mendonça Canamary Jr.1* , Walter Yukihiko Takahashi2 and Juliana Maria Ferraz Sallum1 Canamary Jr. et al. Int J Retin Vitr (2018) 4:1 https://doi.org/10.1186/s40942-017-0104-9 International Journal of Retina and Vitreous REVIEW Open Access Autoimmune retinopathy: A Review Aristófanes Mendonça Canamary Jr.1* , Walter Yukihiko Takahashi2 and Juliana Maria Ferraz Sallum1 Abstract Autoimmune retinopathy (AIR) is a rare and still poorly understood immune-mediated disease that may cause infam- mation from circulating autoantibodies against the retina. It may be related to history of autoimmune disease in the patient or in a family member or the presence of neoplastic disease in the individual. The disease may be subdivided into paraneoplastic and non-paraneoplastic AIR. When related to melanoma, it is referred to as MAR, and when related to other cancers, it is called CAR. The exact prevalence of AIR is unknown. It mainly afects older adults. Patients present with bilateral and asymmetric scotomas, photopsias, visual feld defects, with rapidly progressive visual loss in late onset. In the initial stage, fundus examination is unremarkable, and in late stages, there is limited retinal epithe- liopathy and vascular attenuation, with or without optic disc pallor, associated or not with intraocular infammation and with no evidence of degenerative retinal disease. A clinical investigation with detailed anamnesis and laboratory tests should be performed to search for an associated neoplasm. Ophthalmologic and complementary examina- tions such as full-feld electroretinogram, optical coherence tomography, visual feld and fundus autofuorescence, help the diagnosis. Blood tests to search for autoantibodies should be requested. Management consists of prolonged immunosuppression, which may be combined with antioxidant vitamins. In general, the prognosis is uncertain, so the disease still needs to be better understood. More studies should be performed to improve diagnostic measures and defne specifc management that could preserve or even restore vision. Keywords: Autoantibodies, Recoverin, Immunosuppression, Retinal cone photoreceptor cells, Rod cell outer segment Background Te clinical examination may show some abnormalities Autoimmune retinopathy (AIR) is a rare infammatory such as little or no retinal pigment epitheliopathy, vas- condition that can lead to blindness, and while it has cular attenuation, optic disc pallor, with minimal or no been studied for several years, it still remains under diag- ocular signs of infammation [6]. Although these altera- nosed [1]. It had been associated with an immune-medi- tions have been described, most of the cases have no ated component and is related to circulating antiretinal ocular fndings on fundus examination [7]. History of antibodies, which are believed to be responsible for the cancer, autoimmune diseases in the family, presence of retinopathy [2, 3], although the precise mechanisms are circulating autoantibodies against the retina, progressive not entirely understood. visual loss, associated with alterations in full-feld ERG, Te initial signs and symptoms depend on the type of with the presence or not of the ophthalmologic fndings cell most afected and the antiretinal antibody involved, described above, with no evidence of degenerative eye causing a diverse clinical, anatomical and functional disease, may lead to a diagnosis of AIR [2, 6]. OCT [8], presentation [4]. visual feld, and FAF help the diagnosis, and a search for AIR is characterized by usually bilateral, suddenly pro- an associated or not neoplastic condition needs to be gressive, painless visual deterioration [5], scotomas, vis- performed [9, 10]. Tis retinopathy can be divided in two ual feld defect and retinal (photoreceptor) dysfunction. main forms: presumed non-paraneoplastic AIR (npAIR) and paraneoplastic AIR [11]. Te frst one is not related *Correspondence: [email protected] to cancer and is probably the most common. Te sec- 1 Federal University of São Paulo, Rua Botucatu 821, Vila Clementino, São ond includes cancer-associated retinopathy (CAR) and Paulo, SP CEP: 04023‑062, Brazil melanoma-associated retinopathy (MAR), viteliform Full list of author information is available at the end of the article © The Author(s) 2018. This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/ publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Canamary Jr. et al. Int J Retin Vitr (2018) 4:1 Page 2 of 6 maculopathy, and bilateral difuse uveal melanocytic rod dysfunction secondary to antibodies toward bipolar proliferation. CAR and MAR represent the most com- cells (Table 2) [3]. mon paraneoplastic group (Table 1) [3, 6]. Tough less Photosensitivity, hemeralopia, loss of color vision, and common, paraneoplastic syndromes may afect the optic decreased visual acuity and central vision can be expe- nerve as primary site, a condition known as paraneoplas- rienced with cone dysfunction. Nyctalopia, prolonged tic optic neuropathy (PON) [12]. dark adaptation, and peripheral feld loss are results of Te presence of more than one circulating autoanti- rod malfunction [13]. Photopsia occurs in both cases body in AIR leads to an overlap of clinical features and (Table 3). In early stages, there is good visual acuity, and may even be similar to degenerative retinal disorders [6]. as time goes by, some patients report transient dimming In addition, the lack of standardized diagnostic criteria of vision, which may be mistaken for retinal vascular dis- and its rare incidence make diagnosis even more difcult ease such as Behçet and systemic lupus erythematosus [6]. Appropriate clinical assessment, understanding the [7]. pathophysiology, can help achieve a faster diagnosis, so Te disease is normally bilateral and it can be asymmet- that appropriate management could be started earlier in ric [6]. Minimal or no signs of intraocular infammation the course of the disease, thereby increasing the chance can be seen [6]. Funduscopic fndings at presentation are of preventing or decreasing visual worsening. unremarkable in all forms of AIR [3]. However, changes Te aim of this study was to better understand this may occur over time, including vascular attenuation, dif- disease to achieve an earlier diagnosis and initiate the fuse retinal atrophy, mottling of the retinal pigment epi- appropriate management more quickly, but even so, fur- thelium and occasionally optic disc pallor (Fig. 1) [13]. ther research are needed. In cases of PON, the optic disc can appear normal, edematous or atrophic, accompanied by vitreous cells, Epidemiology and retinal vascular leakage, with bilateral subacute, Te exact prevalence of this disease is unknown. It painless and progressive visual loss in a few days [14]. mainly afects older adults. A review of 209 patients with non-paraneoplastic retinopathy determined an average Diagnosis age at diagnosis of 65 years, where women were the most At the onset of the disease, the absence of clinical fnd- afected [5], although other authors reported a case in a ings makes the diagnosis challenging. However, patients 3-year-old child [7]. Presumed non-paraneoplastic retin- who present with progressive visual loss, with no previ- opathy is the most prevalent of all forms of AIR. MAR ous history, with an apparently normal eye fundus, will seems to afect more men, due to melanoma being more be highly suspected of AIR [3]. prevalent in men, and is believed to be increasing in fre- A thorough assessment of the patient’s visual func- quency relative to CAR. Even so, CAR is currently still tion should be performed. Complementary examinations the most common form of paraneoplastic retinopathy such as visual feld, color vision tests, FAF (Fig. 2), FA, [11]. Te malignancy most frequently associated with OCT, and, in some cases, full-feld ERG can be helpful this disorder is small-cell lung cancer, followed by breast [5]. and gynecologic (uterine, ovarian and cervical) carci- noma. Other cancer associations include hematological, prostate, colon and lymphomas [5, 13]. Table 2 Autoimmune retinopathy and related cell dys- function Clinical features Autoimmune retinopathy Cell dysfunction Patients with npAIR typically present with sudden vision Non-paraneoplastic Cones or rods or both loss, scotomas, photopsias, nyctalopia or photoaversion Paraneoplastic and dyschromatopsia [7]. Signs and symptoms can difer CAR Cones and rods depending on which retinal cells are afected. CAR afects MAR Rods, antibodies against bipolar cells both cones and rods, while MAR characteristically shows CAR cancer-associated retinopathy, MAR melanoma-associated retinopathy Table 1 Types of autoimmune retinopathy Type of autoimmune retinopathy Associated conditions Non-paraneoplastic Not related to cancer Paraneoplastic CAR, MAR, viteliform maculopathy, bilateral difuse uveal melanocytic proliferation Canamary Jr. et al. Int J Retin Vitr (2018) 4:1 Page 3 of 6 Table 3 Retinal cell dysfunction and associated symptoms Retinal cell dysfunction Symptoms Cones Photopsia, photosensitivity, hemeralopia, loss of color vision, diminished vision acuity, diminished central vision Rods Photopsia, nyctalopia, prolonged
Load more

Recommended publications
  • Diagnosis and Treatment of Paraneoplastic Syndromes 1
    Instructions: • Each speaker will prepare a syllabus that must be submitted through the online submission system. • The length of the syllabus will be no shorter than 4 single spaced pages in essay (not point) format, plus references. • Use single spaced, 11 point type and (if possible) Times New Roman font. • When typing the text use word wrap, not hard returns to determine your lines. • If headings and subheadings are used, these may be highlighted by using all caps and bold. • Do not use the header or footer feature or endnotes in preparing the text. • The submission must be submitted online. Title: Diagnosis and Treatment of Paraneoplastic Syndromes Learning Objectives: 1. Describe the spectrum of paraneoplastic syndromes with neuro-ophthalmic features 2. Define the challenges in diagnosis of the paraneoplastic syndromes 3. Explain the therapeutic options for treatment of these diseases CME Questions: 1. The presence of serum antibodies against recoverin a. are pathognomonic for CAR b. are found in the majority of patients with lung cancer c. may be responsible apoptotic cell death in CAR patients d. are best detected by immunofluorescent studies on retina 2. Which of the following is correct regarding therapy for paraneoplastic neuro-ophthalmic disease: a. steroid therapy may be helpful in control of disease b. should be initiated only after there is validation for the presence of autoreactive antibodies c. cytoreduction of the primary tumor is not helpful in controlling the autoimmune component d. biologic immunomodulatory agents have no role in therapy 3. Lambert-Eaton myasthenic syndrome is associated with which of the following: a.
    [Show full text]
  • Autoantibody Profiles and Clinical Association in Thai Patients With
    www.nature.com/scientificreports OPEN Autoantibody profles and clinical association in Thai patients with autoimmune retinopathy Aulia Rahmi Pawestri1,8, Niracha Arjkongharn2,8, Ragkit Suvannaboon2,3,8, Aekkachai Tuekprakhon2,4, Vichien Srimuninnimit5, Suthipol Udompunthurak6, La‑ongsri Atchaneeyasakul2, Ajchara Koolvisoot7* & Adisak Trinavarat2* Autoimmune retinopathy (AIR) is a rare immune‑mediated infammation of the retina. The autoantibodies against retinal proteins and glycolytic enzymes were reported to be involved in the pathogenesis. This retrospective cohort study assessed the antiretinal autoantibody profles and their association with clinical outcomes of AIR patients in Thailand. We included 44 patients, 75% were females, with the overall median age of onset of 48 (17–74, IQR 40–55.5) years. Common clinical presentations were nyctalopia (65.9%), blurred vision (52.3%), constricted visual feld (43.2%), and nonrecordable electroretinography (65.9%). Underlying malignancy and autoimmune diseases were found in 2 and 12 female patients, respectively. We found 41 autoantibodies, with anti‑α‑enolase (65.9%) showing the highest prevalence, followed by anti‑CAII (43.2%), anti‑aldolase (40.9%), and anti‑GAPDH (36.4%). Anti‑aldolase was associated with male gender (P = 0.012, OR 7.11, 95% CI 1.54– 32.91). Anti‑CAII showed signifcant association with age of onset (P = 0.025, 95% CI − 17.28 to − 1.24), while anti‑α‑enolase (P = 0.002, OR 4.37, 95% CI 1.83–10.37) and anti‑GAPDH (P = 0.001, OR 1.87, 95% CI 1.32–2.64) were signifcantly associated with nonrecordable electroretinography. Association between the antibody profles and clinical outcomes may be used to direct and adjust the treatment plans and provide insights in the pathogenesis of AIR.
    [Show full text]
  • Major Review
    SURVEY OF OPHTHALMOLOGY VOLUME 48 • NUMBER 1 • JANUARY–FEBRUARY 2003 MAJOR REVIEW Paraneoplastic Retinopathies and Optic Neuropathies Jane W. Chan, MD Department of Internal Medicine, Division of Neurology, University of Nevada School of Medicine, Las Vegas, Nevada, USA Abstract. Unusual neuro-ophthalmologic symptoms and signs that go unexplained should warrant a thorough investigation for paraneoplastic syndromes. Although these syndromes are rare, these clinical manifestations can herald an unsuspected, underlying malignancy that could be treated early and aggressively. This point underscores the importance of distinguishing and understanding the various, sometimes subtle, presentations of ocular paraneoplastic syndromes. Outlined in this review article are diagnostic features useful in differentiating cancer-associated retinopathy, melanoma-associated retin- opathy, and paraneoplastic optic neuropathy. These must also be distinguished from non–cancer- related eye disorders that may clinically resemble cancer-associated retinopathy. The associated anti- bodies and histopathology of each syndrome are presented to help in the understanding of these autoimmune phenomena. Treatment outcomes from reported cases are summarized, and some poten- tial novel immunotherapies are also discussed. (Surv Ophthalmol 48:12–38, 2003. © 2003 by Else- vier Science Inc. All rights reserved.) Key words. autoimmune • cancer-associated retinopathy • melanocytic-associated retinopathy • optic neuropathy • paraneoplastic I. Cancer-Associated Retinopathy nual percent change (based on rates age-adjusted to the 2000 U.S. standard population) for invasive lung Cancer-associated retinopathy (CAR) has been Ϫ thought to be one of the most common paraneo- and bronchial cancer decreased from 3.0 to 0.9 plastic retinopathies. Its incidence is equal among from 1973 to 1999. During the same time period women and men.
    [Show full text]
  • Consensus on the Diagnosis and Management of Nonparaneoplastic Autoimmune Retinopathy Using a Modified Delphi Approach
    Consensus on the Diagnosis and Management of Nonparaneoplastic Autoimmune Retinopathy Using a Modified Delphi Approach AUSTIN R. FOX, LYNN K. GORDON, JOHN R. HECKENLIVELY, JANET L. DAVIS, DEBRA A. GOLDSTEIN, CAREEN Y. LOWDER, ROBERT B. NUSSENBLATT, NICHOLAS J. BUTLER, MONICA DALAL, THIRAN JAYASUNDERA, WENDY M. SMITH, RICHARD W. LEE, GRAZYNA ADAMUS, CHI-CHAO CHAN, JOHN J. HOOKS, CATHERINE W. MORGANS, BARBARA DETRICK, AND H. NIDA SEN PURPOSE: To develop diagnostic criteria for nonpara- second-line treatments, though a consensus agreed that neoplastic autoimmune retinopathy (AIR) through biologics and intravenous immunoglobulin were consid- expert panel consensus and to examine treatment patterns ered appropriate in the treatment of nonparaneoplastic among clinical experts. AIR patients regardless of the stage of disease. Experts DESIGN: Modified Delphi process. agreed that more evidence is needed to treat nonparaneo- METHODS: A survey of uveitis specialists in the Amer- plastic AIR patients with long-term immunomodulatory ican Uveitis Society, a face-to-face meeting (AIR Work- therapy and that there is enough equipoise to justify ran- shop) held at the National Eye Institute, and 2 iterations domized, placebo-controlled trials to determine if nonpar- of expert panel surveys were used in a modified Delphi aneoplastic AIR patients should be treated with long-term process. The expert panel consisted of 17 experts, immunomodulatory therapy. Regarding antiretinal anti- including uveitis specialists and researchers with exper- body detection, consensus agreed that a standardized tise in antiretinal antibody detection. Supermajority assay system is needed to detect serum antiretinal anti- consensus was used and defined as 75% of experts in bodies. Consensus agreed that an ideal assay should agreement.
    [Show full text]
  • Qt2mh1j0hw.Pdf
    UCLA UCLA Previously Published Works Title Consensus on the Diagnosis and Management of Nonparaneoplastic Autoimmune Retinopathy Using a Modified Delphi Approach. Permalink https://escholarship.org/uc/item/2mh1j0hw Authors Fox, Austin R Gordon, Lynn K Heckenlively, John R et al. Publication Date 2016-08-01 DOI 10.1016/j.ajo.2016.05.013 Peer reviewed eScholarship.org Powered by the California Digital Library University of California Consensus on the Diagnosis and Management of Nonparaneoplastic Autoimmune Retinopathy Using a Modified Delphi Approach AUSTIN R. FOX, LYNN K. GORDON, JOHN R. HECKENLIVELY, JANET L. DAVIS, DEBRA A. GOLDSTEIN, CAREEN Y. LOWDER, ROBERT B. NUSSENBLATT, NICHOLAS J. BUTLER, MONICA DALAL, THIRAN JAYASUNDERA, WENDY M. SMITH, RICHARD W. LEE, GRAZYNA ADAMUS, CHI-CHAO CHAN, JOHN J. HOOKS, CATHERINE W. MORGANS, BARBARA DETRICK, AND H. NIDA SEN PURPOSE: To develop diagnostic criteria for nonpara- second-line treatments, though a consensus agreed that neoplastic autoimmune retinopathy (AIR) through biologics and intravenous immunoglobulin were consid- expert panel consensus and to examine treatment patterns ered appropriate in the treatment of nonparaneoplastic among clinical experts. AIR patients regardless of the stage of disease. Experts DESIGN: Modified Delphi process. agreed that more evidence is needed to treat nonparaneo- METHODS: A survey of uveitis specialists in the Amer- plastic AIR patients with long-term immunomodulatory ican Uveitis Society, a face-to-face meeting (AIR Work- therapy and that there is enough equipoise to justify ran- shop) held at the National Eye Institute, and 2 iterations domized, placebo-controlled trials to determine if nonpar- of expert panel surveys were used in a modified Delphi aneoplastic AIR patients should be treated with long-term process.
    [Show full text]
  • Case Report Intravenous Immunoglobulin for Management Of
    Case Report Intravenous Immunoglobulin for Management of Non-paraneoplastic Autoimmune Retinopathy Sahba Fekri1,2, MD; Masoud Soheilian1,2, MD; Babak Rahimi-Ardabili1, MD 1Ophthalmic Research Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran 2Department of Ophthalmology, Labbafinejad Medical Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran ORCID: Sahba Fekri: https://orcid.org/0000-0002-7388-6725 Abstract Purpose: To report a case of non-paraneoplastic autoimmune retinopathy (npAIR) treated with intravenous immunoglobulin (IVIG). Case report: A 12-year-old boy presented with progressive visual field loss, nyctalopia, and flashing for three months. He had suffered from common cold two weeks before the onset of these symptoms. On the basis of clinical history and paraclinical findings, he was diagnosed with npAIR, and IVIG without immunosuppressive therapy was started. During the one-year follow-up period after the first course of IVIG, flashing disappeared completely. Visual acuity remained 10/10, but nyctalopia did not improve. Multimodal imaging showed no disease progression. Conclusion: Although established retinal degenerative changes seem irreversible in npAIR, IVIG may be a suitable choice to control the disease progression. Keywords: Autoimmune Retinopathy; Intravenous Immunoglobulin; Nyctalopia; Photopsia; Retinal Degeneration J Ophthalmic Vis Res 2020; 15 (2): 246–251 INTRODUCTION retinal disorders, caused by circulating antiretinal antibodies (ARAs). They can be paraneoplastic (pAIR) or, more commonly, non-neoplastic Autoimmune retinopathies (AIRs) are a heteroge- (npAIR) characterized by bilateral, often neous group of immune-mediated degenerative asymmetric, progressive, painless visual acuity or visual field loss over weeks to months with photopsias and scotomas.[1] Depending Correspondence to: on the cell type and antigen targeted by Sahba Fekri, MD.
    [Show full text]
  • Ocular Paraneoplastic Syndromes
    biomedicines Review Ocular Paraneoplastic Syndromes Joanna Prze´zdziecka-Dołyk 1,2, Anna Brzecka 3 , Maria Ejma 4, Marta Misiuk-Hojło 1 , Luis Fernando Torres Solis 5, Arturo Solís Herrera 6, Siva G. Somasundaram 7 , Cecil E. Kirkland 7 and Gjumrakch Aliev 8,9,10,11,* 1 Department of Ophthalmology, Wroclaw Medical University, Borowska 213, 50-556 Wrocław, Poland; [email protected] (J.P.-D.); [email protected] (M.M.-H.) 2 Department of Optics and Photonics, Wrocław University of Science and Technology, Wyspia´nskiego27, 50-370 Wrocław, Poland 3 Department of Pulmonology and Lung Oncology, Wrocław Medical University, Grabiszy´nska 105, 53-439 Wrocław, Poland; [email protected] 4 Department of Neurology, Wroclaw Medical University, Borowska 213, 50-556 Wrocław, Poland; [email protected] 5 The School of Medicine, Universidad Autónoma de Aguascalientes, Aguascalientes 20392, Mexico; [email protected] 6 Human Photosynthesis© Research Centre, Aguascalientes 20000, Mexico; [email protected] 7 Department of Biological Sciences, Salem University, Salem, WV 26426, USA; [email protected] (S.G.S.); [email protected] (C.E.K.) 8 Sechenov First Moscow State Medical University (Sechenov University), St. Trubetskaya, 8, bld. 2, 119991 Moscow, Russia 9 Research Institute of Human Morphology, Russian Academy of Medical Science, Street Tsyurupa 3, 117418 Moscow, Russia 10 Institute of Physiologically Active Compounds, Russian Academy of Sciences, Chernogolovka, 142432 Moscow, Russia 11 GALLY International Research Institute, 7733 Louis Pasteur Drive, #330, San Antonio, TX 78229, USA * Correspondence: [email protected] or [email protected]; Tel.: +1-210-442-8625 or +1-440-263-7461 Received: 6 September 2020; Accepted: 8 November 2020; Published: 10 November 2020 Abstract: Ocular-involving paraneoplastic syndromes present a wide variety of clinical symptoms.
    [Show full text]
  • Outcomes in Autoimmune Retinopathy Patients Treated with Rituximab
    Outcomes in Autoimmune Retinopathy Patients Treated With Rituximab SAMANEH DAVOUDI, NAZANIN EBRAHIMIADIB, CAGLA YASA, DAMLA D. SEVGI, RAMAK ROOHIPOOR, EVANGELIA PAPAVASILIEOU, JASON COMANDER, AND LUCIA SOBRIN PURPOSE: To evaluate clinical and ancillary testing, UTOIMMUNE RETINOPATHY (AIR) RESULTS FROM A including adaptive optics, outcomes in autoimmune presumed immunologic attack on the retina by retinopathy (AIR) patients treated with rituximab. antibodies against retinal antigens. The spectrum A DESIGN: Retrospective, interventional case series. of AIR includes nonparaneoplastic AIR (npAIR), METHODS: PATIENTS: Sixteen AIR patients treated with cancer-associated retinopathy (CAR), melanoma- rituximab. OBSERVATION PROCEDURES: All patients were associated retinopathy (MAR), and autoimmune-related treated with a loading and maintenance dose schedule of retinopathy and optic neuropathy (ARRON). There is intravenous rituximab. Visual acuity (VA), electroreti- no established treatment protocol for AIR, and the nography (ERG), and spectral-domain optical coherence evidence base for therapeutic intervention includes only tomography (SDOCT) and visual field (VF) results a few retrospective case series and case reports.1 The largest were recorded. A subset of patients was also imaged using retrospective series assessed therapeutic effects of immuno- adaptive optics scanning laser ophthalmoscopy suppressive medications including cyclosporine, predni- (AO-SLO). MAIN OUTCOME MEASURES: Rates of VA sone, infliximab, azathioprine, and mycophenolate in 30 change before vs after rituximab initiation were compared consecutive patients with AIR.2 They found potential ben- with mixed-model linear regression. efits from using immunosuppression to treat AIR, but this RESULTS: The rate of visual decline was significantly series did not include any patients treated with rituximab. less after rituximab initiation compared with the rate of Given the proposed antibody-mediated nature of AIR, a visual decline prior to rituximab initiation (P [ .005).
    [Show full text]
  • Autoantibody Against Transient Receptor Potential M1 Cation
    Wang et al. BMC Ophthalmology 2012, 12:56 http://www.biomedcentral.com/1471-2415/12/56 CASE REPORT Open Access Autoantibody against transient receptor potential M1 cation channels of retinal ON bipolar cells in paraneoplastic vitelliform retinopathy Yujuan Wang1,2, Mones S Abu-Asab1, Wei Li3, Mary E Aronow4, Arun D Singh4 and Chi-Chao Chan1* Abstract Background: Paraneoplastic retinopathy is caused by the cross-reaction of neoplasm-directed autoantibodies against retinal antigens and results in retinal damage. Paraneoplastic vitelliform retinopathy, a presumed paraneoplastic retinopathy with features of atypical melanoma-associated retinopathy, has recently been reported in patients with metastatic melanoma. Ocular ultrastructure and its autoantibody localization of paraneoplastic vitelliform retinopathy are still indefinable. This is the first report of anti-transient receptor potential M1 antibody directly against human retinal bipolar dendritic tips in a melanoma patient with paraneoplastic vitelliform retinopathy. Case presentation: We present a pair of postmortem eyes of an 80-year-old male with metastatic cutaneous melanoma, who developed paraneoplastic vitelliform retinopathy. The autopsied eyes were examined with light microscopy, immunohistochemistry, and transmission electron microscopy. Microscopically, the inner nuclear layer and outer plexiform layer were the most affected retinal structures, with local thinning. The lesions extended to the outer nuclear layer, resulting in focal retinal degeneration, edema, and atrophy. No active inflammation or melanoma cells were observed. Immunohistochemistry showed tightly compact bipolar cell nuclei (protein kinase C alpha/calbindin positive) with blur/loss of ON bipolar cell dendritic tips (transient receptor potential M1 positive) in diffusely condensed outer plexiform layer. The metastatic melanoma cells in his lung also showed immunoreactivity against transient receptor potential M1 antibody.
    [Show full text]
  • Title Clinical and Immunological Characterization Of
    View metadata, citation and similar papers at core.ac.uk brought to you by CORE provided by Kyoto University Research Information Repository Clinical and immunological characterization of paraneoplastic Title retinopathy. Makiyama, Yukiko; Kikuchi, Takanobu; Otani, Atsushi; Oishi, Author(s) Akio; Guo, Congrong; Nakagawa, Satoko; Ogino, Ken; Kojima, Hiroshi; Kurimoto, Masafumi; Yoshimura, Nagahisa Investigative ophthalmology & visual science (2013), 54(8): Citation 5424-5431 Issue Date 2013-08 URL http://hdl.handle.net/2433/187393 Right © Association for Research in Vision and Ophthalmology Type Journal Article Textversion author Kyoto University Clinical and Immunological Characterization of Paraneoplastic Retinopathy Short title: Characterization of Paraneoplastic Retinopathy Yukiko Makiyama,1 Takanobu Kikuchi,2 Atsushi Otani,1 Akio Oishi,1 Congrong Guo,1 Satoko Nakagawa,1 Ken Ogino,1 Hiroshi Kojima,1 Masafumi Kurimoto,1 Nagahisa Yoshimura,1 1Department of Ophthalmology, Kyoto University Graduate School of Medicine, Kyoto 606-8507, Japan 2Department of Instrumental Analysis Research Center for Human and Environmental Science, Shinshu Univeisity, Nagano 390-8621, Japan All correspondence should be addressed to: Atsushi Otani, MD, PhD Department of Ophthalmology, Kyoto University Graduate School of Medicine 54-Kawaharacho, Shyogoin, Sakyo-ku, Kyoto 606-8507, Japan E-mail: [email protected], Fax: +81-75-752-0933 Tel: +81-75-751-3248 1 Key words: paraneoplastic retinopathy, spectral domain-optical coherence tomography, Western blot analysis, immunohistochemistory, autoantibody, anti-retinal antibody 2 Abstract Purpose: To report the clinical and immunological characterization of paraneoplastic retinopathy and to investigate the association between spectral domain-optical coherence tomography (SD-OCT) findings and the target of autoantibodies in paraneoplastic retinopathy (PR).
    [Show full text]
  • Immune Cell Infiltration Into the Eye Is Controlled by IL-10 in Recoverin-Induced Autoimmune Retinopathy
    Immune Cell Infiltration into the Eye Is Controlled by IL-10 in Recoverin-Induced Autoimmune Retinopathy This information is current as Enayat Nikoopour, Cheng-mao Lin, Sarah Sheskey, John R. of September 28, 2021. Heckenlively and Steven K. Lundy J Immunol 2019; 202:1057-1068; Prepublished online 11 January 2019; doi: 10.4049/jimmunol.1800574 http://www.jimmunol.org/content/202/4/1057 Downloaded from Supplementary http://www.jimmunol.org/content/suppl/2019/01/10/jimmunol.180057 Material 4.DCSupplemental http://www.jimmunol.org/ References This article cites 39 articles, 4 of which you can access for free at: http://www.jimmunol.org/content/202/4/1057.full#ref-list-1 Why The JI? Submit online. • Rapid Reviews! 30 days* from submission to initial decision by guest on September 28, 2021 • No Triage! Every submission reviewed by practicing scientists • Fast Publication! 4 weeks from acceptance to publication *average Subscription Information about subscribing to The Journal of Immunology is online at: http://jimmunol.org/subscription Permissions Submit copyright permission requests at: http://www.aai.org/About/Publications/JI/copyright.html Email Alerts Receive free email-alerts when new articles cite this article. Sign up at: http://jimmunol.org/alerts The Journal of Immunology is published twice each month by The American Association of Immunologists, Inc., 1451 Rockville Pike, Suite 650, Rockville, MD 20852 Copyright © 2019 by The American Association of Immunologists, Inc. All rights reserved. Print ISSN: 0022-1767 Online ISSN: 1550-6606. The Journal of Immunology Immune Cell Infiltration into the Eye Is Controlled by IL-10 in Recoverin-Induced Autoimmune Retinopathy Enayat Nikoopour,*,†,‡ Cheng-mao Lin,* Sarah Sheskey,* John R.
    [Show full text]
  • Melanoma-Associated Retinopathy a Paraneoplastic Autoimmune Complication
    CLINICAL SCIENCES Melanoma-Associated Retinopathy A Paraneoplastic Autoimmune Complication Ying Lu, MD, PhD; Lin Jia, MS; Shirley He, MD, MS; Mary C. Hurley, MS; Monique J. Leys, MD; Thiran Jayasundera, MD; John R. Heckenlively, MD Objectives: To study 11 patients with melanoma- patients (9 of 11) had a strong family history of autoim- associated retinopathy (MAR) to clarify the reliability of mune disorders. Any type of melanoma (cutaneous, cho- various methods of diagnostic testing, to determine the roidal, ciliary body, or choroidal nevi) may be associ- underlying antigenic retinal proteins, and to study the ated with this paraneoplastic autoimmune reactivity. MAR clinical histories and types of associated melanomas. may precede or follow the diagnosis of melanoma. Pa- tients with MAR have the same antigenic retinal pro- Methods: Clinical data were obtained from patients with teins that have been associated with cancer-associated reti- melanoma who developed marked visual problems. Test- nopathy. In addition, 2 new antigenic retinal proteins, ing included electroretinography, kinetic visual fields, aldolase A and aldolase C, were found. comparative studies of Western blots, and indirect im- munohistologic examination to detect antiretinal anti- Conclusions: There was a high prevalence of positive bodies, as well as proteomic studies to identify underly- family histories of autoimmune disease in patients with ing antigenic retinal proteins. MAR. To confirm the disorder, multiple clinical and se- rum diagnostic techniques (Western blot or indirect im- Results: Patients with MAR typically have rapid onset of photopsias, scotomata, and loss of central or paracen- munohistologic examination) are needed. Two newly ob- tral vision. Ophthalmoscopy seldom shows significant served antigenic retinal proteins, aldolase A and aldolase changes early, but electroretinograms are abnormal.
    [Show full text]