{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,9,8]],"date-time":"2025-09-08T05:32:04Z","timestamp":1757309524231,"version":"3.40.5"},"reference-count":27,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2021,3,30]],"date-time":"2021-03-30T00:00:00Z","timestamp":1617062400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100003246","name":"Nederlandse Organisatie voor Wetenschappelijk Onderzoek","doi-asserted-by":"publisher","award":["016.156.033","016.196.310"],"award-info":[{"award-number":["016.156.033","016.196.310"]}],"id":[{"id":"10.13039\/501100003246","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Ann Clin Transl Neurol"],"published-print":{"date-parts":[[2021,5]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Objective<\/jats:title><jats:p>To prospectively determine the value of optical coherence tomography (OCT) as a surrogate outcome measure for the progression of myelopathy in males with adrenoleukodystrophy.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Retinal nerve fiber layer (RNFL) and ganglion cell layer (GCL) thickness were measured at baseline, 1\u2010 and 2\u2010year follow\u2010up in patients and age\u2010matched controls. We assessed the severity of myelopathy with clinical parameters: Expanded Disability Status Scale (EDSS), Severity Scoring system for Progressive Myelopathy (SSPROM), and timed up\u2010and\u2010go. Linear mixed model analysis was used to compare changes in retinal layer thickness of patients to controls. In addition, we correlated changes in retinal layer thickness with changes in clinical parameters.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Longitudinal data were available for 28 patients and 29 controls. Peripapillary RNFL (pRNFL) thickness decreased significantly in patients compared to controls (\u22121.75\u00b5m, <jats:italic>p<\/jats:italic>\u00a0=\u00a00.001), whereas change in macular GCL and RNFL was not different between groups. Analysis of the symptomatic subgroup showed that, apart from a similar decrease in pRNFL thickness, GCL thickness decreased significantly (\u22120.55\u00a0\u00b5m, <jats:italic>p<\/jats:italic>\u00a0=\u00a00.014). There were moderately strong correlations between changes in retinal layer thickness and changes in clinical parameters of severity of myelopathy.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>This prospective study demonstrates the potential of OCT\u2010measured retinal neurodegeneration as a surrogate outcome measure for the progression of myelopathy in adrenoleukodystrophy. As differences were small, our findings need to be confirmed with longer follow\u2010up and\/or in a larger patient sample.<\/jats:p><\/jats:sec>","DOI":"10.1002\/acn3.51349","type":"journal-article","created":{"date-parts":[[2021,3,30]],"date-time":"2021-03-30T15:28:57Z","timestamp":1617118137000},"page":"1064-1072","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Optical coherence tomography to measure the progression of myelopathy in adrenoleukodystrophy"],"prefix":"10.1002","volume":"8","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3885-2415","authenticated-orcid":false,"given":"Wouter J. C.","family":"van Ballegoij","sequence":"first","affiliation":[{"name":"Department of Paediatric Neurology Amsterdam Leukodystrophy Center Emma Children\u2019s Hospital Amsterdam University Medical Centers University of Amsterdam Amsterdam The Netherlands"},{"name":"Department of Neurology OLVG Hospital Amsterdam The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Irene C.","family":"Huffnagel","sequence":"additional","affiliation":[{"name":"Department of Paediatric Neurology Amsterdam Leukodystrophy Center Emma Children\u2019s Hospital Amsterdam University Medical Centers University of Amsterdam Amsterdam The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Stephanie I. 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