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Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy

PURPOSE: Paraneoplastic retinopathy can be the first manifestation of systemic malignancy. A subset of paraneoplastic retinopathy is characterized by negative-type electroretinography (ERG) without fundus abnormality. Here we describe the multimodal imaging and clinico-pathological correlation of a...

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Autores principales: Chen, Fred K., Chew, Avenell L., Zhang, Dan, Chen, Shang-Chih, Chelva, Enid, Chandrasekera, Erandi, Koay, Eleanor M. H., Forrester, John, McLenachan, Samuel
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5427140/
https://www.ncbi.nlm.nih.gov/pubmed/28382556
http://dx.doi.org/10.1007/s10633-017-9587-9
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author Chen, Fred K.
Chew, Avenell L.
Zhang, Dan
Chen, Shang-Chih
Chelva, Enid
Chandrasekera, Erandi
Koay, Eleanor M. H.
Forrester, John
McLenachan, Samuel
author_facet Chen, Fred K.
Chew, Avenell L.
Zhang, Dan
Chen, Shang-Chih
Chelva, Enid
Chandrasekera, Erandi
Koay, Eleanor M. H.
Forrester, John
McLenachan, Samuel
author_sort Chen, Fred K.
collection PubMed
description PURPOSE: Paraneoplastic retinopathy can be the first manifestation of systemic malignancy. A subset of paraneoplastic retinopathy is characterized by negative-type electroretinography (ERG) without fundus abnormality. Here we describe the multimodal imaging and clinico-pathological correlation of a unique case of acute progressive paravascular placoid neuroretinopathy with suspected retinal depolarizing bipolar cell dysfunction preceding the diagnosis of metastatic small cell carcinoma of the prostate. METHODS: ERG was performed according to the International Society for Clinical Electrophysiology of Vision standards. Imaging modalities included near-infrared reflectance, blue-light autofluorescence, fluorescein and indocyanine green angiographies, spectral domain optical coherence tomography, ultra-widefield colour and green-light autofluorescence imaging, microperimetry and adaptive optics imaging. Patient serum was screened for anti-retinal antibodies using western blotting. Immunostaining and histological analyses were performed on sections from human retinal tissues and a patient prostate biopsy. RESULTS: Serial multimodal retinal imaging, microperimetry and adaptive optics photography demonstrated a paravascular distribution of placoid lesions characterized by hyper-reflectivity within the outer nuclear layer resembling type 2 acute macular neuroretinopathy. There was no visible lesion within the inner nuclear layer despite electronegative-type ERG. Six months later, the patient presented with metastatic small cell carcinoma of the prostate. Tumour cells were immunopositive for glyceraldehyde-3-phosphate dehydrogenase, enolase and recoverin as well as neuroendocrine markers. The patient’s serum reacted to cytoplasmic and nuclear antigens in the prostate biopsy and in human retina. Anti-retinal antibodies against several antigens were detected by both commercial and in-house western blots. CONCLUSIONS: A spectrum of autoreactive anti-retinal antibodies is associated with a unique phenotype of acute progressive paravascular placoid neuroretinopathy resulting in degeneration of photoreceptor cells, inner retinal dysfunction and classic electronegative ERG in paraneoplastic retinopathy. Detailed clinical, functional and immunological phenotyping of paraneoplastic retinopathy illustrated the complex mechanism of paraneoplastic syndrome. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1007/s10633-017-9587-9) contains supplementary material, which is available to authorized users.
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spelling pubmed-54271402017-05-26 Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy Chen, Fred K. Chew, Avenell L. Zhang, Dan Chen, Shang-Chih Chelva, Enid Chandrasekera, Erandi Koay, Eleanor M. H. Forrester, John McLenachan, Samuel Doc Ophthalmol Clinical Case Report PURPOSE: Paraneoplastic retinopathy can be the first manifestation of systemic malignancy. A subset of paraneoplastic retinopathy is characterized by negative-type electroretinography (ERG) without fundus abnormality. Here we describe the multimodal imaging and clinico-pathological correlation of a unique case of acute progressive paravascular placoid neuroretinopathy with suspected retinal depolarizing bipolar cell dysfunction preceding the diagnosis of metastatic small cell carcinoma of the prostate. METHODS: ERG was performed according to the International Society for Clinical Electrophysiology of Vision standards. Imaging modalities included near-infrared reflectance, blue-light autofluorescence, fluorescein and indocyanine green angiographies, spectral domain optical coherence tomography, ultra-widefield colour and green-light autofluorescence imaging, microperimetry and adaptive optics imaging. Patient serum was screened for anti-retinal antibodies using western blotting. Immunostaining and histological analyses were performed on sections from human retinal tissues and a patient prostate biopsy. RESULTS: Serial multimodal retinal imaging, microperimetry and adaptive optics photography demonstrated a paravascular distribution of placoid lesions characterized by hyper-reflectivity within the outer nuclear layer resembling type 2 acute macular neuroretinopathy. There was no visible lesion within the inner nuclear layer despite electronegative-type ERG. Six months later, the patient presented with metastatic small cell carcinoma of the prostate. Tumour cells were immunopositive for glyceraldehyde-3-phosphate dehydrogenase, enolase and recoverin as well as neuroendocrine markers. The patient’s serum reacted to cytoplasmic and nuclear antigens in the prostate biopsy and in human retina. Anti-retinal antibodies against several antigens were detected by both commercial and in-house western blots. CONCLUSIONS: A spectrum of autoreactive anti-retinal antibodies is associated with a unique phenotype of acute progressive paravascular placoid neuroretinopathy resulting in degeneration of photoreceptor cells, inner retinal dysfunction and classic electronegative ERG in paraneoplastic retinopathy. Detailed clinical, functional and immunological phenotyping of paraneoplastic retinopathy illustrated the complex mechanism of paraneoplastic syndrome. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1007/s10633-017-9587-9) contains supplementary material, which is available to authorized users. Springer Berlin Heidelberg 2017-04-05 2017 /pmc/articles/PMC5427140/ /pubmed/28382556 http://dx.doi.org/10.1007/s10633-017-9587-9 Text en © The Author(s) 2017 Open AccessThis 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.
spellingShingle Clinical Case Report
Chen, Fred K.
Chew, Avenell L.
Zhang, Dan
Chen, Shang-Chih
Chelva, Enid
Chandrasekera, Erandi
Koay, Eleanor M. H.
Forrester, John
McLenachan, Samuel
Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
title Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
title_full Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
title_fullStr Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
title_full_unstemmed Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
title_short Acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
title_sort acute progressive paravascular placoid neuroretinopathy with negative-type electroretinography in paraneoplastic retinopathy
topic Clinical Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5427140/
https://www.ncbi.nlm.nih.gov/pubmed/28382556
http://dx.doi.org/10.1007/s10633-017-9587-9
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