Corticosteroid Treatment for Melanoma-Associated Retinopathy

Jacobzone, C.; Cochard-Marianowski, C.; Kupfer, I.; Bettembourg, S.; Dordain, Y.; Miséry, Laurent; Cochener, Béatrice; Sassolas, B.

doi:10.1001/archderm.140.10.1258

Cited by 25 publications

(4 citation statements)

References 18 publications

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“…In patients with PNR, the therapy of the underlying tumour usually comprises chemotherapy, radiotherapy, and/or surgery 53 , 56 , 57 . A first-line therapy with local or systemic steroids and/or antimetabolites is widely accepted 28 , 33 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 , 66 . The application of IV methylprednisolone has been found to be of more benefit at initiation of therapy than oral prednisone 56 , 61 , 67 .…”

Section: Discussionmentioning

confidence: 99%

Electrodiagnostic Biomarkers in Paraneoplastic Retinopathy

Gougoulakis,

Rothermundt,

Flynn

et al. 2024

Klin Monbl Augenheilkd

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Objective Paraneoplastic retinopathy (PNR) is a rapid-onset photoreceptor and post-photoreceptor dysfunction triggered by a cross-reaction between antigens expressed by the underlying tumour and retinal proteins. The present study aims to determine the electrodiagnostic biomarkers that support the diagnosis of PNR and evaluate the effect of treatment. Methods A retrospective observational case-controlled study including 25 patients with suspected PNR, of which 11 patients were diagnosed with PNR. The presence of PNR was confirmed based on clinical examination, supported by colour fundus photography, fundus autofluorescence imaging, optical coherence tomography, fluorescein angiography, retinal vessel oximetry, colour test, full-field electroretinogram (ffERG), on-/off ERG, S-cone ERG, and multifocal ERG (mfERG). The relationships between the clinical symptomatology and the effect of therapy were evaluated. Results All PNR patients (Nr: 11) presented with subjective symptoms of newly reported central vision or visual field deterioration. Posterior segment findings showed a severe patchy-like retinal atrophy, attenuation of the retinal vessels, and a waxy optic disc. Optical coherence tomography revealed a discontinued ISe line, and multiple hyperreflective foci. Retinal vessel oxygen saturation was increased. Multifocal ERG revealed reduced central and paracentral responses and ffERG severely attenuated scotopic-, photopic-, on-/off- and S-cone responses. The colour vision test revealed a tritan-tetartan-weakness. Two of the PNR patients underwent rituximab therapy with no further progression and even recovery of electrodiagnostic responses.In 1 nPNR (non-paraneoplastic retinopathy) patient (total Nr: 14) pseudoxanthoma elasticum-related retinopathy was the reason for impaired vision. In 3 of 13 patients with bronchopulmonary cancer a MEK- and FGFR-inhibitor- drug toxicity was the reason for the visual deterioration. Conclusion Careful investigation for signs of central and/or peripheral visual field deterioration must be performed in the presence of history of a co-existing malignancy. The possibility of PNR should be taken into account. The electrodiagnostic biomarkers, suggested in this study, may help to promptly recognise PNR and also to evaluate the effect of implemented therapy.

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Section: Discussionmentioning

confidence: 99%

Electrodiagnostic Biomarkers in Paraneoplastic Retinopathy

Gougoulakis,

Rothermundt,

Flynn

et al. 2024

Klin Monbl Augenheilkd

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“…16 Subhadra et al 17 demonstrated improvement in visual field testing after IVIG and Jacobzone et al 18 showed some recovery in ERG after systemic corticosteroids, although this is not typical as treatment of visual loss in MAR has been largely ineffective. Additionally, there is some concern that immunomodulatory therapy such as IVIG, although decreasing the titer of circulating autoantibodies, may increase cancer mortality because MAR patients may have antibodies that are protective against tumor spread.…”

Section: Discussionmentioning

confidence: 99%

Diagnosis of Occult Melanoma Using Transient Receptor Potential Melastatin 1 (TRPM1) Autoantibody Testing

et al. 2013

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Purpose To report the first case of melanoma-associated retinopathy (MAR) and underlying occult melanoma diagnosed based on the presence of serum transient receptor potential melastatin 1 (TRPM1) autoantibodies. Design Interventional case report with basic science correlation. Participants One patient with melanoma-associated retinopathy (MAR). Intervention Testing for the presence of serum TRPM1 autoantibodies. Main Outcome Measure Diagnosis of an occult melanoma involving the axillary lymph nodes (unknown primary) and MAR based on the presence of TRPM1 autoantibodies in the patient’s serum. Results The patient's clinical exam was remarkable for mild intraocular inflammation in both eyes and retinal hemorrhages with an apparent choroidal neovascularization (CNV) in the left eye, which was confirmed by fluorescein angiogram (FA) and indocyanine green angiography (ICG) testing. Humphrey visual field (HVF) demonstrated diffuse depression in both eyes out of proportion to the clinical exam prompting electroretinogram (ERG) testing which revealed an electronegative response. Dark-adapted thresholds were markedly elevated and mediated by cones. Due to concern for MAR, a systemic work-up for melanoma was performed by the primary care physician which was unrevealing. Given our continued clinical suspicion for MAR, the patient's serum was sent for evaluation for TRPM1 autoantibodies. The patient’s serum applied to normal human retina exhibited positivity in the inner nuclear layer. Application of the patient’s serum to wild-type and TRPM1 knockout mouse retina revealed strongly labeled bipolar cells in the wild-type retina, but not in the TRPM1 knockout retina indicating TRPM1-dependent immunoreactivity. The antigen was confirmed as TRPM1 by labeling of TRPM1-transfected human embryonic kidney 293 (HEK293) cells. Additional systemic work-up prompted by this finding resulted in identification of an occult metastatic melanoma involving the axillary lymph nodes with an unknown primary. The patient underwent surgical excision of his occult melanoma without evidence of other sites of metastases. He also received intravenous immunoglobulin therapy and his vision has stabilized. Conclusions This is the first reported case of a melanoma-associated retinopathy diagnosed utilizing the application of innovative approach by testing for serum TRPM1 autoantibodies.

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“…74 That said, reports on visual function usually found progressive deterioration in most patients despite treatment of primary malignancy supplemented with local and systemic corticosteroids. [75][76][77][78][79][80] This prompted explorations of other therapies including high-dose intravenous corticosteroids, corticosteroids plus immunomodulating agents, systemic azathioprine, systemic cyclosporine, systemic mycophenolate, plasmapheresis, intravenous immunoglobulins, and several different monoclonal antibodies (eg, alemtuzumab, rituximab, etc.). The results have been mixed in terms of shortterm outcomes.…”

Section: Therapymentioning

confidence: 99%

Ocular Paraneoplastic Syndromes: A Critical Review of Diffuse Uveal Melanocytic Proliferation and Autoimmune Retinopathy

et al. 2022

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Background: Dozens of paraneoplastic syndromes affect the visual system ranging from conjunctival pemphigoid to encephalopathy of the occipital cortex. The most profiled ocular syndromes are bilateral diffuse uveal melanocytic proliferation (BDUMP) and the autoimmune retinopathies. Purpose: To review the critical features of these 2 entities then concentrate on advancements in treatment made within the last 10 years. Study Design: Literature review with structured data abstraction. Results: Major insights into pathogenesis have been wanting. Plasmapheresis appears to improve vision in a substantial proportion of patients with BDUMP. The number of clinical variables that influence visual outcome in paraneoplastic retinopathies combined with the variety of local and systemic treatment options makes interpretation of clinical effectiveness difficult. Conclusions: The rarity of these disorders makes randomized clinical trials unlikely. It may be time for a clinical professional organization to use a modified Delphi method to establish a consensus algorithm for the diagnosis and management of retinal paraneoplastic syndromes to augment clinical communications and clinical trials.

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Corticosteroid Treatment for Melanoma-Associated Retinopathy

Cited by 25 publications

References 18 publications

Electrodiagnostic Biomarkers in Paraneoplastic Retinopathy

Electrodiagnostic Biomarkers in Paraneoplastic Retinopathy

Diagnosis of Occult Melanoma Using Transient Receptor Potential Melastatin 1 (TRPM1) Autoantibody Testing

Ocular Paraneoplastic Syndromes: A Critical Review of Diffuse Uveal Melanocytic Proliferation and Autoimmune Retinopathy

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