Long-term multimodal imaging in acute posterior multifocal placoid pigment epitheliopathy and association with coxsackievirus exposure
The purpose of this study was to evaluate potential insights into the pathogenesis of acute posterior multifocal placoid pigment epitheliopathy (APMPPE) using multimodal diagnostic imaging and laboratory evaluation in long-term follow-up. A retrospective, single-center case series was conducted on s...
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description | The purpose of this study was to evaluate potential insights into the pathogenesis of acute posterior multifocal placoid pigment epitheliopathy (APMPPE) using multimodal diagnostic imaging and laboratory evaluation in long-term follow-up. A retrospective, single-center case series was conducted on seven consecutive patients (14 eyes) who were given a diagnosis of APMPPE from March 1, 2011, through June 30, 2019 with at least three months of follow-up. Clinical characteristics (age, symptoms, visual acuity [VA]), laboratory testing including coxsackievirus titers, and multimodal imaging from fundus photography, spectral-domain optical coherence tomography (SD-OCT), fundus autofluorescence (FAF), fluorescein angiography (FA), and indocyanine green angiography (ICG) were analyzed for each patient. The initial median VA was 20/71 and final median VA was 20/22. Coxsackievirus B (CVB) titers were elevated ([greater than or equal to] 1:80) in six of seven patients, with a four-fold increase in convalescent titers seen in two patients suggestive of recent infection. All patients were treated with oral corticosteroids, and five patients underwent corticosteroid-sparing immunomodulatory therapy. Initially, multifocal deep choroidal lesions were observed in the posterior pole corresponding to patches of hypocyanescence on ICG. Overlying retinal pigment epithelium (RPE) disease was observed on FAF, although this finding was not universally observed, suggesting that RPE disease may occur as a sequelae to unchecked choroidal inflammation. SD-OCT architectural changes confirmed outer retina and ellipsoid zone disruption. FA of active lesions showed early hypofluorescence and late hyperfluorescence with surrounding leakage while inactive disease showed areas of staining. Long-term follow-up of multimodal diagnostic imaging in APMPPE revealed that choroidal inflammation likely precedes RPE change and photoreceptor damage. Elevation of coxsackievirus titers with seroconversion may be associated with an infectious trigger in concert with immune-mediated disease in this posterior uveitis syndrome. |
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A retrospective, single-center case series was conducted on seven consecutive patients (14 eyes) who were given a diagnosis of APMPPE from March 1, 2011, through June 30, 2019 with at least three months of follow-up. Clinical characteristics (age, symptoms, visual acuity [VA]), laboratory testing including coxsackievirus titers, and multimodal imaging from fundus photography, spectral-domain optical coherence tomography (SD-OCT), fundus autofluorescence (FAF), fluorescein angiography (FA), and indocyanine green angiography (ICG) were analyzed for each patient. The initial median VA was 20/71 and final median VA was 20/22. Coxsackievirus B (CVB) titers were elevated ([greater than or equal to] 1:80) in six of seven patients, with a four-fold increase in convalescent titers seen in two patients suggestive of recent infection. All patients were treated with oral corticosteroids, and five patients underwent corticosteroid-sparing immunomodulatory therapy. Initially, multifocal deep choroidal lesions were observed in the posterior pole corresponding to patches of hypocyanescence on ICG. Overlying retinal pigment epithelium (RPE) disease was observed on FAF, although this finding was not universally observed, suggesting that RPE disease may occur as a sequelae to unchecked choroidal inflammation. SD-OCT architectural changes confirmed outer retina and ellipsoid zone disruption. FA of active lesions showed early hypofluorescence and late hyperfluorescence with surrounding leakage while inactive disease showed areas of staining. Long-term follow-up of multimodal diagnostic imaging in APMPPE revealed that choroidal inflammation likely precedes RPE change and photoreceptor damage. Elevation of coxsackievirus titers with seroconversion may be associated with an infectious trigger in concert with immune-mediated disease in this posterior uveitis syndrome.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0238080</identifier><identifier>PMID: 32834009</identifier><language>eng</language><publisher>San Francisco: Public Library of Science</publisher><subject>Acuity ; Angiography ; Antibodies ; Biology and Life Sciences ; Complications ; Corticoids ; Corticosteroids ; Coxsackievirus infections ; Coxsackieviruses ; Diagnosis ; Diagnostic imaging ; Diagnostic systems ; Epithelium ; Eye (anatomy) ; Fluorescein ; Immunomodulation ; Infections ; Laboratories ; Laboratory tests ; Lesions ; Medical imaging ; Medicine and Health Sciences ; Methods ; Optical Coherence Tomography ; Pathogenesis ; Patients ; Photography ; Pigments ; Research and Analysis Methods ; Retina ; Retinal pigment epithelium ; Risk factors ; Seroconversion ; Signs and symptoms ; Social Sciences ; Supervision ; Surgery ; Uveitis ; Visual acuity</subject><ispartof>PloS one, 2020-08, Vol.15 (8), p.e0238080</ispartof><rights>COPYRIGHT 2020 Public Library of Science</rights><rights>2020 Li et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. 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A retrospective, single-center case series was conducted on seven consecutive patients (14 eyes) who were given a diagnosis of APMPPE from March 1, 2011, through June 30, 2019 with at least three months of follow-up. Clinical characteristics (age, symptoms, visual acuity [VA]), laboratory testing including coxsackievirus titers, and multimodal imaging from fundus photography, spectral-domain optical coherence tomography (SD-OCT), fundus autofluorescence (FAF), fluorescein angiography (FA), and indocyanine green angiography (ICG) were analyzed for each patient. The initial median VA was 20/71 and final median VA was 20/22. Coxsackievirus B (CVB) titers were elevated ([greater than or equal to] 1:80) in six of seven patients, with a four-fold increase in convalescent titers seen in two patients suggestive of recent infection. All patients were treated with oral corticosteroids, and five patients underwent corticosteroid-sparing immunomodulatory therapy. Initially, multifocal deep choroidal lesions were observed in the posterior pole corresponding to patches of hypocyanescence on ICG. Overlying retinal pigment epithelium (RPE) disease was observed on FAF, although this finding was not universally observed, suggesting that RPE disease may occur as a sequelae to unchecked choroidal inflammation. SD-OCT architectural changes confirmed outer retina and ellipsoid zone disruption. FA of active lesions showed early hypofluorescence and late hyperfluorescence with surrounding leakage while inactive disease showed areas of staining. Long-term follow-up of multimodal diagnostic imaging in APMPPE revealed that choroidal inflammation likely precedes RPE change and photoreceptor damage. Elevation of coxsackievirus titers with seroconversion may be associated with an infectious trigger in concert with immune-mediated disease in this posterior uveitis syndrome.</description><subject>Acuity</subject><subject>Angiography</subject><subject>Antibodies</subject><subject>Biology and Life Sciences</subject><subject>Complications</subject><subject>Corticoids</subject><subject>Corticosteroids</subject><subject>Coxsackievirus infections</subject><subject>Coxsackieviruses</subject><subject>Diagnosis</subject><subject>Diagnostic imaging</subject><subject>Diagnostic systems</subject><subject>Epithelium</subject><subject>Eye (anatomy)</subject><subject>Fluorescein</subject><subject>Immunomodulation</subject><subject>Infections</subject><subject>Laboratories</subject><subject>Laboratory tests</subject><subject>Lesions</subject><subject>Medical imaging</subject><subject>Medicine and Health Sciences</subject><subject>Methods</subject><subject>Optical Coherence Tomography</subject><subject>Pathogenesis</subject><subject>Patients</subject><subject>Photography</subject><subject>Pigments</subject><subject>Research and Analysis Methods</subject><subject>Retina</subject><subject>Retinal pigment epithelium</subject><subject>Risk factors</subject><subject>Seroconversion</subject><subject>Signs and symptoms</subject><subject>Social Sciences</subject><subject>Supervision</subject><subject>Surgery</subject><subject>Uveitis</subject><subject>Visual 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multimodal imaging in acute posterior multifocal placoid pigment epitheliopathy and association with coxsackievirus exposure</title><author>Li, Alexa L ; Palejwala, Neal V ; Shantha, Jessica G ; O'Keefe, Ghazala ; Lee, Cecilia S ; Albini, Thomas ; Yeh, Steven</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c669t-84a1e161a196bcad4e41de7abd1f867d16e025e6169c2e4e64b5146227fe70863</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Acuity</topic><topic>Angiography</topic><topic>Antibodies</topic><topic>Biology and Life Sciences</topic><topic>Complications</topic><topic>Corticoids</topic><topic>Corticosteroids</topic><topic>Coxsackievirus infections</topic><topic>Coxsackieviruses</topic><topic>Diagnosis</topic><topic>Diagnostic imaging</topic><topic>Diagnostic systems</topic><topic>Epithelium</topic><topic>Eye 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association with coxsackievirus exposure</atitle><jtitle>PloS one</jtitle><date>2020-08-24</date><risdate>2020</risdate><volume>15</volume><issue>8</issue><spage>e0238080</spage><pages>e0238080-</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>The purpose of this study was to evaluate potential insights into the pathogenesis of acute posterior multifocal placoid pigment epitheliopathy (APMPPE) using multimodal diagnostic imaging and laboratory evaluation in long-term follow-up. A retrospective, single-center case series was conducted on seven consecutive patients (14 eyes) who were given a diagnosis of APMPPE from March 1, 2011, through June 30, 2019 with at least three months of follow-up. Clinical characteristics (age, symptoms, visual acuity [VA]), laboratory testing including coxsackievirus titers, and multimodal imaging from fundus photography, spectral-domain optical coherence tomography (SD-OCT), fundus autofluorescence (FAF), fluorescein angiography (FA), and indocyanine green angiography (ICG) were analyzed for each patient. The initial median VA was 20/71 and final median VA was 20/22. Coxsackievirus B (CVB) titers were elevated ([greater than or equal to] 1:80) in six of seven patients, with a four-fold increase in convalescent titers seen in two patients suggestive of recent infection. All patients were treated with oral corticosteroids, and five patients underwent corticosteroid-sparing immunomodulatory therapy. Initially, multifocal deep choroidal lesions were observed in the posterior pole corresponding to patches of hypocyanescence on ICG. Overlying retinal pigment epithelium (RPE) disease was observed on FAF, although this finding was not universally observed, suggesting that RPE disease may occur as a sequelae to unchecked choroidal inflammation. SD-OCT architectural changes confirmed outer retina and ellipsoid zone disruption. FA of active lesions showed early hypofluorescence and late hyperfluorescence with surrounding leakage while inactive disease showed areas of staining. Long-term follow-up of multimodal diagnostic imaging in APMPPE revealed that choroidal inflammation likely precedes RPE change and photoreceptor damage. Elevation of coxsackievirus titers with seroconversion may be associated with an infectious trigger in concert with immune-mediated disease in this posterior uveitis syndrome.</abstract><cop>San Francisco</cop><pub>Public Library of Science</pub><pmid>32834009</pmid><doi>10.1371/journal.pone.0238080</doi><tpages>e0238080</tpages><orcidid>https://orcid.org/0000-0003-1994-7213</orcidid><orcidid>https://orcid.org/0000-0002-5235-6741</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Acuity Angiography Antibodies Biology and Life Sciences Complications Corticoids Corticosteroids Coxsackievirus infections Coxsackieviruses Diagnosis Diagnostic imaging Diagnostic systems Epithelium Eye (anatomy) Fluorescein Immunomodulation Infections Laboratories Laboratory tests Lesions Medical imaging Medicine and Health Sciences Methods Optical Coherence Tomography Pathogenesis Patients Photography Pigments Research and Analysis Methods Retina Retinal pigment epithelium Risk factors Seroconversion Signs and symptoms Social Sciences Supervision Surgery Uveitis Visual acuity |
title | Long-term multimodal imaging in acute posterior multifocal placoid pigment epitheliopathy and association with coxsackievirus exposure |
url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-25T07%3A30%3A24IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-gale_plos_&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Long-term%20multimodal%20imaging%20in%20acute%20posterior%20multifocal%20placoid%20pigment%20epitheliopathy%20and%20association%20with%20coxsackievirus%20exposure&rft.jtitle=PloS%20one&rft.au=Li,%20Alexa%20L&rft.date=2020-08-24&rft.volume=15&rft.issue=8&rft.spage=e0238080&rft.pages=e0238080-&rft.issn=1932-6203&rft.eissn=1932-6203&rft_id=info:doi/10.1371/journal.pone.0238080&rft_dat=%3Cgale_plos_%3EA633404666%3C/gale_plos_%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2436840106&rft_id=info:pmid/32834009&rft_galeid=A633404666&rft_doaj_id=oai_doaj_org_article_013bf7a4fc2448cc8dd6a002260a58d0&rfr_iscdi=true |