Characteristic retinal atrophy pattern allows differentiation between pediatric MOGAD and MS after a single optic neuritis episode

Background Optic neuritis (ON) is the most prevalent manifestation of pediatric multiple sclerosis (MS ped ) and myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD ped ) in children > 6 years. In this study, we investigated retinal atrophy patterns and diagnostic accuracy of o...

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Veröffentlicht in:Journal of neurology 2022-12, Vol.269 (12), p.6366-6376
Hauptverfasser: Pakeerathan, T., Havla, J., Schwake, C., Salmen, A., Bigi, S., Abegg, M., Brügger, D., Ferrazzini, T., Runge, A.-K., Breu, M., Kornek, B., Bsteh, G., Felipe-Rucián, A., Ringelstein, M., Aktas, O., Karenfort, M., Wendel, E., Kleiter, I., Hellwig, K., Kümpfel, T., Thiels, C., Lücke, T., Gold, R., Rostasy, K., Ayzenberg, I.
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container_end_page 6376
container_issue 12
container_start_page 6366
container_title Journal of neurology
container_volume 269
creator Pakeerathan, T.
Havla, J.
Schwake, C.
Salmen, A.
Bigi, S.
Abegg, M.
Brügger, D.
Ferrazzini, T.
Runge, A.-K.
Breu, M.
Kornek, B.
Bsteh, G.
Felipe-Rucián, A.
Ringelstein, M.
Aktas, O.
Karenfort, M.
Wendel, E.
Kleiter, I.
Hellwig, K.
Kümpfel, T.
Thiels, C.
Lücke, T.
Gold, R.
Rostasy, K.
Ayzenberg, I.
description Background Optic neuritis (ON) is the most prevalent manifestation of pediatric multiple sclerosis (MS ped ) and myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD ped ) in children > 6 years. In this study, we investigated retinal atrophy patterns and diagnostic accuracy of optical coherence tomography (OCT) in differentiating between both diseases after the first ON episode. Methods Patients were retrospectively identified in eight tertial referral centers. OCT, VEP and high/low-contrast visual acuity (HCVA/LCVA) have been investigated > 6 months after the first ON. Prevalence of pathological OCT findings was identified based on data of 144 age-matched healthy controls. Results Thirteen MOGAD ped (10.7 ± 4.2 years, F:M 8:5, 21 ON eyes) and 21 MS ped (14.3 ± 2.4 years, F:M 19:2, 24 ON eyes) patients were recruited. We observed a significantly more profound atrophy of both peripapillary and macular retinal nerve fiber layer in MOGAD ped compared to MS ped (pRNFL global: 68.2 ± 16.9 vs. 89.4 ± 12.3 µm, p  
doi_str_mv 10.1007/s00415-022-11256-y
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In this study, we investigated retinal atrophy patterns and diagnostic accuracy of optical coherence tomography (OCT) in differentiating between both diseases after the first ON episode. Methods Patients were retrospectively identified in eight tertial referral centers. OCT, VEP and high/low-contrast visual acuity (HCVA/LCVA) have been investigated &gt; 6 months after the first ON. Prevalence of pathological OCT findings was identified based on data of 144 age-matched healthy controls. Results Thirteen MOGAD ped (10.7 ± 4.2 years, F:M 8:5, 21 ON eyes) and 21 MS ped (14.3 ± 2.4 years, F:M 19:2, 24 ON eyes) patients were recruited. We observed a significantly more profound atrophy of both peripapillary and macular retinal nerve fiber layer in MOGAD ped compared to MS ped (pRNFL global: 68.2 ± 16.9 vs. 89.4 ± 12.3 µm, p  &lt; 0.001; mRNFL: 0.12 ± 0.01 vs. 0.14 ± 0.01 mm 3 , p  &lt; 0.001). Neither other macular layers nor P100 latency differed. MOGAD ped developed global atrophy affecting all peripapillary segments, while MS ped displayed predominantly temporal thinning. Nasal pRNFL allowed differentiation between both diseases with the highest diagnostic accuracy (AUC = 0.902, cutoff &lt; 62.5 µm, 90.5% sensitivity and 70.8% specificity for MOGAD ped ). OCT was also substantially more sensitive compared to VEP in identification of ON eyes in MOGAD (pathological findings in 90% vs. 14%, p  = 0.016). Conclusion First MOGAD-ON results in a more severe global peripapillary atrophy compared to predominantly temporal thinning in MS-ON. Nasal pRNFL allows differentiation between both diseases with the highest accuracy, supporting the additional diagnostic value of OCT in children with ON.</description><identifier>ISSN: 0340-5354</identifier><identifier>ISSN: 1432-1459</identifier><identifier>EISSN: 1432-1459</identifier><identifier>DOI: 10.1007/s00415-022-11256-y</identifier><identifier>PMID: 35869995</identifier><language>eng</language><publisher>Berlin/Heidelberg: Springer Berlin Heidelberg</publisher><subject>Accuracy ; Acuity ; Atrophy ; Atrophy - pathology ; Eye ; Humans ; Latency ; Medicine ; Medicine &amp; Public Health ; Multiple sclerosis ; Multiple Sclerosis - complications ; Myelin ; Neuritis ; Neurology ; Neuroradiology ; Neurosciences ; Oligodendrocyte-myelin glycoprotein ; Optic neuritis ; Optic Neuritis - diagnosis ; Original Communication ; Patients ; Pediatrics ; Retina ; Retina - diagnostic imaging ; Retina - pathology ; Retinal degeneration ; Retinal Degeneration - pathology ; Retrospective Studies ; Thinning ; Tomography, Optical Coherence - methods ; Vision Disorders ; Visual evoked potentials</subject><ispartof>Journal of neurology, 2022-12, Vol.269 (12), p.6366-6376</ispartof><rights>The Author(s) 2022</rights><rights>2022. The Author(s).</rights><rights>The Author(s) 2022. This work is published under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c474t-6284e83679e7798182c6dcf6830a34040093a22f090f635504e676702164f3dd3</citedby><cites>FETCH-LOGICAL-c474t-6284e83679e7798182c6dcf6830a34040093a22f090f635504e676702164f3dd3</cites><orcidid>0000-0002-6009-792X</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s00415-022-11256-y$$EPDF$$P50$$Gspringer$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s00415-022-11256-y$$EHTML$$P50$$Gspringer$$Hfree_for_read</linktohtml><link.rule.ids>230,314,776,780,881,27901,27902,41464,42533,51294</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/35869995$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Pakeerathan, T.</creatorcontrib><creatorcontrib>Havla, J.</creatorcontrib><creatorcontrib>Schwake, C.</creatorcontrib><creatorcontrib>Salmen, A.</creatorcontrib><creatorcontrib>Bigi, S.</creatorcontrib><creatorcontrib>Abegg, M.</creatorcontrib><creatorcontrib>Brügger, D.</creatorcontrib><creatorcontrib>Ferrazzini, T.</creatorcontrib><creatorcontrib>Runge, A.-K.</creatorcontrib><creatorcontrib>Breu, M.</creatorcontrib><creatorcontrib>Kornek, B.</creatorcontrib><creatorcontrib>Bsteh, G.</creatorcontrib><creatorcontrib>Felipe-Rucián, A.</creatorcontrib><creatorcontrib>Ringelstein, M.</creatorcontrib><creatorcontrib>Aktas, O.</creatorcontrib><creatorcontrib>Karenfort, M.</creatorcontrib><creatorcontrib>Wendel, E.</creatorcontrib><creatorcontrib>Kleiter, I.</creatorcontrib><creatorcontrib>Hellwig, K.</creatorcontrib><creatorcontrib>Kümpfel, T.</creatorcontrib><creatorcontrib>Thiels, C.</creatorcontrib><creatorcontrib>Lücke, T.</creatorcontrib><creatorcontrib>Gold, R.</creatorcontrib><creatorcontrib>Rostasy, K.</creatorcontrib><creatorcontrib>Ayzenberg, I.</creatorcontrib><title>Characteristic retinal atrophy pattern allows differentiation between pediatric MOGAD and MS after a single optic neuritis episode</title><title>Journal of neurology</title><addtitle>J Neurol</addtitle><addtitle>J Neurol</addtitle><description>Background Optic neuritis (ON) is the most prevalent manifestation of pediatric multiple sclerosis (MS ped ) and myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD ped ) in children &gt; 6 years. In this study, we investigated retinal atrophy patterns and diagnostic accuracy of optical coherence tomography (OCT) in differentiating between both diseases after the first ON episode. Methods Patients were retrospectively identified in eight tertial referral centers. OCT, VEP and high/low-contrast visual acuity (HCVA/LCVA) have been investigated &gt; 6 months after the first ON. Prevalence of pathological OCT findings was identified based on data of 144 age-matched healthy controls. Results Thirteen MOGAD ped (10.7 ± 4.2 years, F:M 8:5, 21 ON eyes) and 21 MS ped (14.3 ± 2.4 years, F:M 19:2, 24 ON eyes) patients were recruited. We observed a significantly more profound atrophy of both peripapillary and macular retinal nerve fiber layer in MOGAD ped compared to MS ped (pRNFL global: 68.2 ± 16.9 vs. 89.4 ± 12.3 µm, p  &lt; 0.001; mRNFL: 0.12 ± 0.01 vs. 0.14 ± 0.01 mm 3 , p  &lt; 0.001). Neither other macular layers nor P100 latency differed. MOGAD ped developed global atrophy affecting all peripapillary segments, while MS ped displayed predominantly temporal thinning. Nasal pRNFL allowed differentiation between both diseases with the highest diagnostic accuracy (AUC = 0.902, cutoff &lt; 62.5 µm, 90.5% sensitivity and 70.8% specificity for MOGAD ped ). OCT was also substantially more sensitive compared to VEP in identification of ON eyes in MOGAD (pathological findings in 90% vs. 14%, p  = 0.016). Conclusion First MOGAD-ON results in a more severe global peripapillary atrophy compared to predominantly temporal thinning in MS-ON. Nasal pRNFL allows differentiation between both diseases with the highest accuracy, supporting the additional diagnostic value of OCT in children with ON.</description><subject>Accuracy</subject><subject>Acuity</subject><subject>Atrophy</subject><subject>Atrophy - pathology</subject><subject>Eye</subject><subject>Humans</subject><subject>Latency</subject><subject>Medicine</subject><subject>Medicine &amp; Public Health</subject><subject>Multiple sclerosis</subject><subject>Multiple Sclerosis - complications</subject><subject>Myelin</subject><subject>Neuritis</subject><subject>Neurology</subject><subject>Neuroradiology</subject><subject>Neurosciences</subject><subject>Oligodendrocyte-myelin glycoprotein</subject><subject>Optic neuritis</subject><subject>Optic Neuritis - diagnosis</subject><subject>Original Communication</subject><subject>Patients</subject><subject>Pediatrics</subject><subject>Retina</subject><subject>Retina - diagnostic imaging</subject><subject>Retina - pathology</subject><subject>Retinal degeneration</subject><subject>Retinal Degeneration - pathology</subject><subject>Retrospective Studies</subject><subject>Thinning</subject><subject>Tomography, Optical Coherence - methods</subject><subject>Vision Disorders</subject><subject>Visual evoked potentials</subject><issn>0340-5354</issn><issn>1432-1459</issn><issn>1432-1459</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>C6C</sourceid><sourceid>EIF</sourceid><sourceid>BENPR</sourceid><recordid>eNp9kTtvFDEUhS0EIkvgD1AgSzQ0A34_GqRogYCUKAVQW87MnV1Hs_ZgexJtyy_HYUN4FFSWfb577r0-CD2n5DUlRL8phAgqO8JYRymTqts_QCsqeLsKaR-iFeGCdJJLcYSelHJFCDFNeIyOuDTKWitX6Pt667PvK-RQauhxhhqin7CvOc3bPZ59bVrEfprSTcFDGEfIEGvwNaSIL6HeAEQ8w9BecjM4vzg9eYd9HPD5Z-zHVow9LiFuJsBpvm0RYcmhhoJhDiUN8BQ9Gv1U4NndeYy-fnj_Zf2xO7s4_bQ-Oet6oUXtFDMCDFfagtbWUMN6NfSjMpz4tqcgxHLP2EgsGRWXkghQWmnCqBIjHwZ-jN4efOflcgdD37bIfnJzDjuf9y754P5WYti6Tbp2VlEjmWoGr-4Mcvq2QKluF0oP0-QjpKU4pizXhmphG_ryH_QqLbl9bKM0J1Qba0yj2IHqcyolw3g_DCXuNmJ3iNi1iN3PiN2-Fb34c437kl-ZNoAfgNKkuIH8u_d_bH8AaKSziA</recordid><startdate>20221201</startdate><enddate>20221201</enddate><creator>Pakeerathan, T.</creator><creator>Havla, J.</creator><creator>Schwake, C.</creator><creator>Salmen, A.</creator><creator>Bigi, S.</creator><creator>Abegg, M.</creator><creator>Brügger, D.</creator><creator>Ferrazzini, T.</creator><creator>Runge, A.-K.</creator><creator>Breu, M.</creator><creator>Kornek, B.</creator><creator>Bsteh, G.</creator><creator>Felipe-Rucián, A.</creator><creator>Ringelstein, M.</creator><creator>Aktas, O.</creator><creator>Karenfort, M.</creator><creator>Wendel, E.</creator><creator>Kleiter, I.</creator><creator>Hellwig, K.</creator><creator>Kümpfel, T.</creator><creator>Thiels, C.</creator><creator>Lücke, T.</creator><creator>Gold, R.</creator><creator>Rostasy, K.</creator><creator>Ayzenberg, I.</creator><general>Springer Berlin Heidelberg</general><general>Springer Nature B.V</general><scope>C6C</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7TK</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-6009-792X</orcidid></search><sort><creationdate>20221201</creationdate><title>Characteristic retinal atrophy pattern allows differentiation between pediatric MOGAD and MS after a single optic neuritis episode</title><author>Pakeerathan, T. ; Havla, J. ; Schwake, C. ; Salmen, A. ; Bigi, S. ; Abegg, M. ; Brügger, D. ; Ferrazzini, T. ; Runge, A.-K. ; Breu, M. ; Kornek, B. ; Bsteh, G. ; Felipe-Rucián, A. ; Ringelstein, M. ; Aktas, O. ; Karenfort, M. ; Wendel, E. ; Kleiter, I. ; Hellwig, K. ; Kümpfel, T. ; Thiels, C. ; Lücke, T. ; Gold, R. ; Rostasy, K. ; Ayzenberg, I.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c474t-6284e83679e7798182c6dcf6830a34040093a22f090f635504e676702164f3dd3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Accuracy</topic><topic>Acuity</topic><topic>Atrophy</topic><topic>Atrophy - pathology</topic><topic>Eye</topic><topic>Humans</topic><topic>Latency</topic><topic>Medicine</topic><topic>Medicine &amp; Public Health</topic><topic>Multiple sclerosis</topic><topic>Multiple Sclerosis - complications</topic><topic>Myelin</topic><topic>Neuritis</topic><topic>Neurology</topic><topic>Neuroradiology</topic><topic>Neurosciences</topic><topic>Oligodendrocyte-myelin glycoprotein</topic><topic>Optic neuritis</topic><topic>Optic Neuritis - diagnosis</topic><topic>Original Communication</topic><topic>Patients</topic><topic>Pediatrics</topic><topic>Retina</topic><topic>Retina - diagnostic imaging</topic><topic>Retina - pathology</topic><topic>Retinal degeneration</topic><topic>Retinal Degeneration - pathology</topic><topic>Retrospective Studies</topic><topic>Thinning</topic><topic>Tomography, Optical Coherence - methods</topic><topic>Vision Disorders</topic><topic>Visual evoked potentials</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Pakeerathan, T.</creatorcontrib><creatorcontrib>Havla, J.</creatorcontrib><creatorcontrib>Schwake, C.</creatorcontrib><creatorcontrib>Salmen, A.</creatorcontrib><creatorcontrib>Bigi, S.</creatorcontrib><creatorcontrib>Abegg, M.</creatorcontrib><creatorcontrib>Brügger, D.</creatorcontrib><creatorcontrib>Ferrazzini, T.</creatorcontrib><creatorcontrib>Runge, A.-K.</creatorcontrib><creatorcontrib>Breu, M.</creatorcontrib><creatorcontrib>Kornek, B.</creatorcontrib><creatorcontrib>Bsteh, G.</creatorcontrib><creatorcontrib>Felipe-Rucián, A.</creatorcontrib><creatorcontrib>Ringelstein, M.</creatorcontrib><creatorcontrib>Aktas, O.</creatorcontrib><creatorcontrib>Karenfort, M.</creatorcontrib><creatorcontrib>Wendel, E.</creatorcontrib><creatorcontrib>Kleiter, I.</creatorcontrib><creatorcontrib>Hellwig, K.</creatorcontrib><creatorcontrib>Kümpfel, T.</creatorcontrib><creatorcontrib>Thiels, C.</creatorcontrib><creatorcontrib>Lücke, T.</creatorcontrib><creatorcontrib>Gold, R.</creatorcontrib><creatorcontrib>Rostasy, K.</creatorcontrib><creatorcontrib>Ayzenberg, I.</creatorcontrib><collection>Springer Nature OA Free Journals</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Neurosciences Abstracts</collection><collection>Health &amp; 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In this study, we investigated retinal atrophy patterns and diagnostic accuracy of optical coherence tomography (OCT) in differentiating between both diseases after the first ON episode. Methods Patients were retrospectively identified in eight tertial referral centers. OCT, VEP and high/low-contrast visual acuity (HCVA/LCVA) have been investigated &gt; 6 months after the first ON. Prevalence of pathological OCT findings was identified based on data of 144 age-matched healthy controls. Results Thirteen MOGAD ped (10.7 ± 4.2 years, F:M 8:5, 21 ON eyes) and 21 MS ped (14.3 ± 2.4 years, F:M 19:2, 24 ON eyes) patients were recruited. We observed a significantly more profound atrophy of both peripapillary and macular retinal nerve fiber layer in MOGAD ped compared to MS ped (pRNFL global: 68.2 ± 16.9 vs. 89.4 ± 12.3 µm, p  &lt; 0.001; mRNFL: 0.12 ± 0.01 vs. 0.14 ± 0.01 mm 3 , p  &lt; 0.001). Neither other macular layers nor P100 latency differed. MOGAD ped developed global atrophy affecting all peripapillary segments, while MS ped displayed predominantly temporal thinning. Nasal pRNFL allowed differentiation between both diseases with the highest diagnostic accuracy (AUC = 0.902, cutoff &lt; 62.5 µm, 90.5% sensitivity and 70.8% specificity for MOGAD ped ). OCT was also substantially more sensitive compared to VEP in identification of ON eyes in MOGAD (pathological findings in 90% vs. 14%, p  = 0.016). Conclusion First MOGAD-ON results in a more severe global peripapillary atrophy compared to predominantly temporal thinning in MS-ON. Nasal pRNFL allows differentiation between both diseases with the highest accuracy, supporting the additional diagnostic value of OCT in children with ON.</abstract><cop>Berlin/Heidelberg</cop><pub>Springer Berlin Heidelberg</pub><pmid>35869995</pmid><doi>10.1007/s00415-022-11256-y</doi><tpages>11</tpages><orcidid>https://orcid.org/0000-0002-6009-792X</orcidid><oa>free_for_read</oa></addata></record>
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source MEDLINE; Springer Nature - Complete Springer Journals
subjects Accuracy
Acuity
Atrophy
Atrophy - pathology
Eye
Humans
Latency
Medicine
Medicine & Public Health
Multiple sclerosis
Multiple Sclerosis - complications
Myelin
Neuritis
Neurology
Neuroradiology
Neurosciences
Oligodendrocyte-myelin glycoprotein
Optic neuritis
Optic Neuritis - diagnosis
Original Communication
Patients
Pediatrics
Retina
Retina - diagnostic imaging
Retina - pathology
Retinal degeneration
Retinal Degeneration - pathology
Retrospective Studies
Thinning
Tomography, Optical Coherence - methods
Vision Disorders
Visual evoked potentials
title Characteristic retinal atrophy pattern allows differentiation between pediatric MOGAD and MS after a single optic neuritis episode
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