{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T23:36:57Z","timestamp":1791329817512,"version":"4.3.1"},"reference-count":22,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2016,1,19]],"date-time":"2016-01-19T00:00:00Z","timestamp":1453161600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2016,7]]},"abstract":"Aim<\/jats:title>Brain magnetic resonance imaging (MRI) motor development score (MDS<\/jats:styled-content>) correlations were used to analyze the natural time\u2010course of hypomyelinating PLP<\/jats:styled-content>1<\/jats:italic>\u2010related disorders (Pelizaeus\u2010Merzbacher disease [PMD] and spastic paraplegia type 2).<\/jats:p><\/jats:sec>Method<\/jats:title>Thirty\u2010five male patients (ranging from 0.7\u201343.5y at the first MRI<\/jats:styled-content>) with PLP<\/jats:styled-content>1<\/jats:italic>\u2010related disorder were prospectively followed over 7 years. Patients were classified according to best motor function acquired before 5 years (MDS<\/jats:styled-content>) into five categories (from PMD<\/jats:styled-content>0 without motor acquisition to PMD<\/jats:styled-content>4 with autonomous walking). We determined myelination and atrophy scores and measured corpus callosum area, volume of cerebellum, white matter and grey matter on 63 MRI<\/jats:styled-content>.<\/jats:p><\/jats:sec>Results<\/jats:title>Age\u2010adjusted multivariate analysis revealed that patients with PMD<\/jats:styled-content>0\u20101 had higher\u2010severity atrophy scores and smaller corpus callosum area than did patients with PMD<\/jats:styled-content>2 and PMD<\/jats:styled-content>3\u20104. Myelination score increased until 12 years. There was evidence that the mean myelination differed in frontal white matter, arcuate fibres, and internal capsules among the groups. Most patients showed worsening atrophy (brain, cerebellum, corpus callosum), whereas grey matter and white matter proportions did not change.<\/jats:p><\/jats:sec>Interpretation<\/jats:title>Brain atrophy and myelination of anterior cerebral regions appear to be pertinent biomarkers of motor development. The time\u2010course of inter\u2010 and intra\u2010individual cerebral white matter and grey matter atrophy suggests that both oligodendrocytes and neurons are involved in the physiopathology of PLP<\/jats:styled-content>1<\/jats:italic>\u2010related disorders.<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.13025","type":"journal-article","created":{"date-parts":[[2016,1,20]],"date-time":"2016-01-20T04:28:42Z","timestamp":1453264122000},"page":"706-713","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":28,"title":["Time\u2010course of myelination and atrophy on cerebral imaging in 35 patients with PLP<\/scp>1<\/i>\u2010related disorders"],"prefix":"10.1111","volume":"58","author":[{"given":"Catherine","family":"Sarret","sequence":"first","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"},{"name":"Department of Paediatrics Clermont\u2010Ferrand University Hospital Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean\u2010Jacques","family":"Lemaire","sequence":"additional","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"},{"name":"Department of Neurosurgery Clermont\u2010Ferrand University Hospital Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Davide","family":"Tonduti","sequence":"additional","affiliation":[{"name":"Inserm U1141 Paris Diderot Sorbonne University\u2013Paris Cit\u00e9 DHU PROTECT Robert Debr\u00e9 Hospital Paris France"},{"name":"Department of Child Neurology Neurological Institute C. Besta Foundation IRCCS Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Sontheimer","sequence":"additional","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3262-0867","authenticated-orcid":false,"given":"Jerome","family":"Coste","sequence":"additional","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bruno","family":"Pereira","sequence":"additional","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"},{"name":"Biostatistics Unit (DRCI) Clermont\u2010Ferrand University Hospital Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fabien","family":"Feschet","sequence":"additional","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Basile","family":"Roche","sequence":"additional","affiliation":[{"name":"Image\u2010Guided Clinical Neuroscience and Connectomics (IGCNC) Clermont University University of Auvergne Clermont\u2010Ferrand France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Odile","family":"Boespflug\u2010Tanguy","sequence":"additional","affiliation":[{"name":"Inserm U1141 Paris Diderot Sorbonne University\u2013Paris Cit\u00e9 DHU PROTECT Robert Debr\u00e9 Hospital Paris France"},{"name":"Department of Child Neurology and Metabolic Diseases Leukodystrophies Reference Centre Robert Debr\u00e9 Hospital Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2016,1,19]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ejhg.5200537"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awq257"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1007\/978-3-662-03078-3_4"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0887-8994(97)00212-9"},{"key":"e_1_2_7_6_1","first-page":"1822","article-title":"MR\u2010revealed myelination in the cerebral corticospinal tract as a marker for Pelizaeus\u2010Merzbacher's disease with proteolipid protein gene duplication","volume":"20","author":"Takanashi J","year":"1999","journal-title":"Am J Neuroradiol"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-2003-41276"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1126\/scitranslmed.3004373"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11910-008-0034-x"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jns.2013.08.030"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10048-004-0207-y"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/00001648-199001000-00010"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2288-2-8"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/0887-8994(94)00124-K"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12154"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00401-009-0562-8"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awf043"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.23-06-02265.2003"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1002\/glia.20325"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-9861(19980518)394:4<506::AID-CNE8>3.0.CO;2-5"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.19-19-08349.1999"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroscience.2009.12.047"},{"key":"e_1_2_7_23_1","first-page":"65","article-title":"Determination of cerebellar volume in children and adolescents with magnetic resonance images","volume":"71","author":"Kosar MI","year":"2012","journal-title":"Folia Morphol"}],"container-title":["Developmental Medicine & Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fdmcn.13025","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.13025","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,23]],"date-time":"2023-09-23T14:37:44Z","timestamp":1695479864000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/dmcn.13025"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,1,19]]},"references-count":22,"journal-issue":{"issue":"7","published-print":{"date-parts":[[2016,7]]}},"alternative-id":["10.1111\/dmcn.13025"],"URL":"https:\/\/doi.org\/10.1111\/dmcn.13025","archive":["Portico"],"relation":{},"ISSN":["0012-1622","1469-8749"],"issn-type":[{"value":"0012-1622","type":"print"},{"value":"1469-8749","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,1,19]]},"assertion":[{"value":"2015-11-08","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2016-01-19","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}