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Retinal Cases & Brief Reports Nov 2022Melanoma-associated retinopathy responds poorly to currently-available therapies, with continued chronic decline in visual function being the norm, despite treatment....
PURPOSE
Melanoma-associated retinopathy responds poorly to currently-available therapies, with continued chronic decline in visual function being the norm, despite treatment. The purpose of this report is to describe the excellent response of a patient with melanoma-associated retinopathy to a triple therapy regimen of rituximab, intravenous immunoglobulin, and intravitreal corticosteroids.
METHODS
Single interventional case report describing management of melanoma-associated retinopathy and the patient's response to this treatment. Retinal function was monitored by serial visual acuity, fundus exams, Goldmann visual fields, and electroretinography.
RESULTS
A 65-year old man presented with new onset photopsia, decrease visual acuity and nyctalopia in both eyes in the setting of recently-diagnosed Stage IIIB melanoma, initially treated with wide local excision and adjuvant interferon. He was diagnosed with melanoma-associated retinopathy that initially worsened during his course of interferon for treatment of the melanoma. We initiated triple therapy with rituximab, intravenous immunoglobulin, and intravitreal corticosteroids, and this resulted in full return of electroretinography function and resumption of 20/20 visual acuity in both eyes.
CONCLUSION
This is the first reported case of the utility of triple therapy with rituximab, intravenous immunoglobulin, and intravitreal steroids for successful management of melanoma-associated retinopathy as demonstrated by improvement in acuity, symptoms, visual fields, and electroretinography.
Topics: Male; Humans; Aged; Paraneoplastic Syndromes, Ocular; Immunoglobulins, Intravenous; Rituximab; Electroretinography; Melanoma; Adrenal Cortex Hormones; Steroids; Interferons
PubMed: 33165302
DOI: 10.1097/ICB.0000000000001081 -
Retinal Cases & Brief Reports Sep 2021To describe a case of choroidal neovascularization (CNV) and chorioretinal scarring in a patient with melanoma-associated retinopathy after ipilimumab/nivolumab...
PURPOSE
To describe a case of choroidal neovascularization (CNV) and chorioretinal scarring in a patient with melanoma-associated retinopathy after ipilimumab/nivolumab combination immune therapy for malignant melanoma.
METHODS
Retrospective case report with fundus photography, fluorescein angiography, optical coherence tomography, and electroretinography.
RESULTS
A 65-year-old woman presented with symptoms of photopsia and visual field loss. She had previously undergone ipilimumab/nivolumab combination chemotherapy treatment for malignant melanoma 14 months earlier coinciding with the onset of her visual symptoms. Fundus photography showed bilateral atrophic chorioretinal lesions and peripheral retinal pigment epithelial changes. Fluorescein angiography revealed retinovascular leakage in both eyes with CNV in the right eye. Optical coherence tomography showed a pigment epithelial detachment with subretinal fluid and subretinal hyperreflective material consistent with occult CNV. Visual field testing showed generalized visual field loss in both eyes. Bloodwork discovered an elevated angiotensin-converting enzyme. Electroretinography revealed abnormal peripheral rod and cone function with impairment of the photoreceptor and inner nuclear layer. Serum Western blot was positive for 60 kDa antiretinal autoantibody. After a single bevacizumab injection in the right eye, CNV resolved and visual acuity improved from 20/50 before the injection to 20/25 3 months after the injection. Visual acuity in the left eye deteriorated for months to counting fingers but then improved to 20/100 on follow-up examinations.
CONCLUSION
Ipilimumab and nivolumab have been associated with immune-related ocular adverse effects. We report a case of combination therapy presenting with chorioretinal scarring and subsequent CNV in a patient with melanoma-associated retinopathy, a rare yet important adverse effect.
Topics: Aged; Choroid Diseases; Choroidal Neovascularization; Drug Therapy, Combination; Female; Fluorescein Angiography; Humans; Ipilimumab; Nivolumab; Paraneoplastic Syndromes, Ocular; Retrospective Studies; Tomography, Optical Coherence
PubMed: 31246886
DOI: 10.1097/ICB.0000000000000882 -
American Journal of Ophthalmology Case... Mar 2023To report a case of acute posterior multifocal placoid pigment epitheliopathy (APMPPE) following COVID-19 infection.
PURPOSE
To report a case of acute posterior multifocal placoid pigment epitheliopathy (APMPPE) following COVID-19 infection.
OBSERVATIONS
A 17-year-old female developed central scotomas and photopsias two weeks after SARS-CoV-2 diagnosis with polymerase chain reaction studies. She presented with poor visual acuity of 3/60 on the Feinbloom eye chart in the left eye. Dilated examination and multi-modal retinal imaging were consistent with the diagnosis of APMPPE, with noteworthy subretinal fluid. The patient was treated with an oral prednisone taper starting at 60mg with rapid resolution in subretinal fluid and improvement of visual acuity. Five weeks after presentation, visual acuity improved to 20/20 OU with complete resolution of the creamy white choroidal lesions and subretinal fluid.
CONCLUSION
There is a growing body of literature reporting the ocular manifestations of COVID-19. Rarely inflammation of the retina or choroid have been associated with the infection. To the best of our knowledge, there are no prior reports that describe the clinical course or visual outcome in a patient with APMPEE associated with recent COVID-19 infection. Accordingly, we are not aware of any other reports that describe the treatment of APMPEE associated with COVID-19 with corticosteroids. The mechanism linking COVID-19 infection to inflammatory ocular disorders in unclear and likely multi-factorial.
PubMed: 36597447
DOI: 10.1016/j.ajoc.2022.101790 -
Retinal Cases & Brief Reports Jul 2022To report a case of acute zonal occult outer retinopathy in which adaptive optics (AO) facilitated visualization of abnormal photoreceptors previously thought to be in...
PURPOSE
To report a case of acute zonal occult outer retinopathy in which adaptive optics (AO) facilitated visualization of abnormal photoreceptors previously thought to be in an area of normal retina on conventional optical coherence tomography (OCT).
METHODS
Case report.
RESULTS
A 51-year-old woman presents with 11-month history of photopsias and scotoma in the temporal visual field of her left eye. Ocular imaging including fluorescein angiography, fundus autofluorescence and OCT suggested the diagnosis of acute zonal occult outer retinopathy in the left eye. Adaptive optics optical coherence tomography (AO-OCT) revealed photoreceptor abnormalities not previously identified in conventional OCT, in areas apparently normal on multimodal imaging. On enface and cross-sectional AO-OCT, round and evenly spaced hyperreflectivity corresponding to normal cone mosaic (Pattern 1) was adjacent to unevenly and disrupted cone hyperreflectivity (Pattern 2) and areas with hyporeflectivity or no cone reflectivity (Pattern 3). Cross-sectional AO-OCT of Patterns 2 and 3 also revealed attenuation of ellipsoid zone with loss of interdigitation zone.
CONCLUSION
Adaptive optics OCT documented cone photoreceptors in finer details than conventional OCT and revealed early changes in a patient with acute zonal occult outer retinopathy, in an area of the retina thought to be normal on conventional multimodal imaging. These findings may provide important insight into pathogenesis and progression of the disease.
Topics: Cross-Sectional Studies; Female; Fluorescein Angiography; Humans; Middle Aged; Scotoma; Tomography, Optical Coherence; Visual Acuity; White Dot Syndromes
PubMed: 32271274
DOI: 10.1097/ICB.0000000000001000 -
Journal of Ophthalmic & Vision Research 2020To report a case of non-paraneoplastic autoimmune retinopathy (npAIR) treated with intravenous immunoglobulin (IVIG).
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.
PubMed: 32308960
DOI: 10.18502/jovr.v15i2.6743 -
American Journal of Ophthalmology Case... Jun 2022To report a case of a branch retinal vein occlusion (BRVO) following mRNA COVID-19 vaccination.
PURPOSE
To report a case of a branch retinal vein occlusion (BRVO) following mRNA COVID-19 vaccination.
OBSERVATIONS
A 34-year-old healthy male presented with blurriness in the inferior visual field, intermittent photopsia, multiple retinal hemorrhages, dilated and tortuous retinal vessels, and cotton wools spots in the right eye. The clinical examination and ancillary tests confirmed the diagnosis of a right eye BRVO. The visual symptoms started 2 days following first dose COVID-19 vaccination with the BNT162b2 (Pfizer-BioNTech) mRNA vaccine.
CONCLUSIONS AND IMPORTANCE
This is a rare case of BRVO in an otherwise healthy young man, presenting after vaccination for COVID-19 in the absence of other coagulable risk factors. As the literature on venous thrombosis after COVID-19 vaccinations remains sparse, it is critical to raise awareness that BRVO could be a vaccine-related thrombotic adverse event. We highlight that as more of the population is vaccinated, an increased incidence of BRVO may confirm the link to COVID-19 vaccination.
PubMed: 35211659
DOI: 10.1016/j.ajoc.2022.101445 -
Retinal Cases & Brief Reports Jul 2023To report a case of nonparaneoplastic autoimmune retinopathy with phenotypical features of pericentral retinal degeneration (PRD) who responded to IV immunoglobulin...
PURPOSE
To report a case of nonparaneoplastic autoimmune retinopathy with phenotypical features of pericentral retinal degeneration (PRD) who responded to IV immunoglobulin therapy.
METHODS
A case report. A 27-year-old man presented with recent subacute progressive nyctalopia and photopsia.
RESULTS
Dilated fundoscopy demonstrated confluent yellow-white patches along the main temporal vascular arcades with sparing of the central island in the posterior pole. Color vision, fundus autofluorescence, fluorescein angiography, static visual field, and electroretinographic studies were inconclusive for retinal degeneration. Subsequent genetic testing for known mutations was negative. Workup for paraneoplastic autoimmune retinopathy was negative. Antiretinal antibodies were positive. The patient was diagnosed with nonparaneoplastic autoimmune retinopathy and was treated with IV immunoglobulin, which resulted in objective and subjective improvement on electroretinography, visual field, and optical coherence tomography of the retina.
CONCLUSION
Nonparaneoplastic autoimmune retinopathy may present in a patient with the clinical phenotype of PRD. It is essential to rule out nonparaneoplastic autoimmune retinopathy in patients with subacute changes in the natural course of pericentral retinal degeneration because treatment with IV immunoglobulin may be helpful.
Topics: Humans; Retinal Diseases; Retinal Degeneration; Autoimmune Diseases; Immunoglobulins, Intravenous; Retina; Electroretinography; Paraneoplastic Syndromes; Phenotype; Tomography, Optical Coherence; Fluorescein Angiography
PubMed: 34618714
DOI: 10.1097/ICB.0000000000001199 -
GROWTH OF PRESUMED CHOROIDAL NEVUS INTO MELANOMA OVER 4 YEARS IN BAP1 TUMOR PREDISPOSITION SYNDROME.Retinal Cases & Brief Reports Mar 2021To report a case of presumed choroidal nevus that eventually grew into melanoma in a patient with family history of choroidal melanoma and germline BAP1 mutation.
PURPOSE
To report a case of presumed choroidal nevus that eventually grew into melanoma in a patient with family history of choroidal melanoma and germline BAP1 mutation.
METHODS
Case report.
RESULTS
A 55-year-old healthy white woman with a family history of uveal melanoma in her father, paternal aunt, and paternal cousin was referred for evaluation of an asymptomatic small pigmented choroidal lesion in her right eye, measuring 2 mm × 2 mm in basal diameter and 1 mm in thickness. There were no clinical risk factors. The patient was advised routine monitoring but returned 4 years later with intermittent photopsia. The choroidal mass demonstrated growth and suggestive of transformation into melanoma, measuring 9 mm × 6 mm in basal diameter and 2.5 mm in thickness with overlying orange lipofuscin pigment and no associated subretinal fluid. Fine-needle aspiration biopsy disclosed Chromosome 3 mosaic monosomy and Chromosomes 6 and 8 disomy. Iodine 125 plaque radiotherapy was provided. Based on growth to melanoma and strong family history of uveal melanoma, BAP1 germline mutation testing was performed, and the results were positive.
CONCLUSION
This case demonstrates growth of a presumed choroidal nevus into melanoma in the setting of underlying germline BAP1 mutation. We suggest that small pigmented choroidal lesions be monitored closely in patients with germline BAP1 mutation or with family history of uveal melanoma, even in the absence of known local risk factors predictive of tumor growth.
Topics: Biopsy, Needle; Cell Transformation, Neoplastic; Choroid Neoplasms; Chromosomes, Human, Pair 3; Female; Genetic Predisposition to Disease; Genetic Techniques; Germ-Line Mutation; Humans; Melanoma; Middle Aged; Nevus, Pigmented; Tumor Suppressor Proteins; Ubiquitin Thiolesterase
PubMed: 29994997
DOI: 10.1097/ICB.0000000000000772 -
BMJ Open Ophthalmology 2022To demonstrate the spectrum of autoimmune retinopathy (AIR) associated with immunotherapy for advanced cutaneous melanoma.
Autoimmune retinopathy with associated anti-retinal antibodies as a potential immune-related adverse event associated with immunotherapy in patients with advanced cutaneous melanoma: case series and systematic review.
OBJECTIVE
To demonstrate the spectrum of autoimmune retinopathy (AIR) associated with immunotherapy for advanced cutaneous melanoma.
METHODS AND ANALYSIS
Retrospective chart review on patients with advanced cutaneous melanoma who developed AIR after initiating immunotherapy. Complete ophthalmic examination and relevant ancillary testing were performed on each patient. The presence of AIR-associated anti-retinal antibodies was confirmed by western blot and/or immunohistochemical staining. Ophthalmic and systemic outcomes after treatment for AIR were followed over time. A systematic review of AIR associated with immunotherapy for cutaneous or non-ocular mucosal melanoma was carried out in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines.
RESULTS
Case 1 developed photopsia and nyctalopia with electroretinographic findings characteristic for melanoma-associated retinopathy 1 week after initiating ipilimumab/nivolumab immunotherapy. Case 2 experienced new severe bilateral visual field loss associated with anti-retinal and anti-optic nerve antibodies while on maintenance nivolumab immunotherapy. Case 3 developed decreased visual acuity due to acute exudative polymorphous vitelliform maculopathy within 2 weeks of initiating ipilimumab/nivolumab immunotherapy. All patients had concurrent extraocular immune-related adverse events in addition to the presence of anti-retinal antibodies on serological testing. 14 published cases of AIR associated with immunotherapy for cutaneous or non-ocular mucosal melanoma were identified and reviewed.
CONCLUSIONS
Immune checkpoint inhibition can trigger the development of AIR with varied clinical manifestations in patients with advanced cutaneous melanoma. This study highlights the need for close monitoring in cutaneous melanoma patients receiving immunotherapy who develop new visual symptoms with or without funduscopic changes, as well as the potential role for screening of patients prior to initiating immunotherapy.
Topics: Antibodies, Monoclonal, Humanized; Autoimmune Diseases; Humans; Immunologic Factors; Immunotherapy; Ipilimumab; Melanoma; Nivolumab; Retinal Diseases; Retrospective Studies; Skin Neoplasms; Melanoma, Cutaneous Malignant
PubMed: 35047671
DOI: 10.1136/bmjophth-2021-000889 -
Journal of Neuro-ophthalmology : the... Dec 2021A 58-year-old man noticed shadows in the temporal visual field of each eye and photopsias within these scotomas for the past year. Formal visual fields demonstrated...
A 58-year-old man noticed shadows in the temporal visual field of each eye and photopsias within these scotomas for the past year. Formal visual fields demonstrated bitemporal hemianopia. MRI of the sella was normal, but fundus autofluorescence (FAF) demonstrated an area of hyperautofluorescence in the nasal retina of each eye corresponding to the loss of retinal pigment epithelium and photoreceptors. A diagnosis of acute zonal occult outer retinopathy was made based on the presence of trizonal involvement on FAF. This case is a reminder that bitemporal hemianopia can be caused by a retinopathy involving nasal retina in each eye, and FAF is a very useful test when assessing patients with suspected outer retinal disorders.
Topics: Fluorescein Angiography; Fundus Oculi; Hemianopsia; Humans; Male; Middle Aged; Scotoma; Tomography, Optical Coherence; Visual Acuity; White Dot Syndromes
PubMed: 33470740
DOI: 10.1097/WNO.0000000000001167