{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T02:33:23Z","timestamp":1790649203327,"version":"4.1.0"},"reference-count":42,"publisher":"Oxford University Press (OUP)","issue":"7","license":[{"start":{"date-parts":[[2022,12,13]],"date-time":"2022-12-13T00:00:00Z","timestamp":1670889600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100007660","name":"University of Antwerp","doi-asserted-by":"publisher","award":["38694"],"award-info":[{"award-number":["38694"]}],"id":[{"id":"10.13039\/501100007660","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007393","name":"Association Fran\u00e7aise contre les Myopathies","doi-asserted-by":"publisher","award":["24063"],"award-info":[{"award-number":["24063"]}],"id":[{"id":"10.13039\/100007393","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100012220","name":"Hercules Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100012220","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100016171","name":"Institute Born-Bunge","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100016171","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003130","name":"FWO","doi-asserted-by":"publisher","award":["41425"],"award-info":[{"award-number":["41425"]}],"id":[{"id":"10.13039\/501100003130","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002661","name":"Flanders Fund for Scientific Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002661","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003130","name":"Research Fund\u2014Flanders","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003130","id-type":"DOI","asserted-by":"publisher"}]},{"name":"European Reference Network for Rare Neuromuscular Diseases"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,7,3]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Charcot\u2013Marie\u2013Tooth disease is the most common inherited disorder of the PNS. CMT1A accounts for 40\u201350% of all cases and is caused by a duplication of the PMP22 gene on chromosome 17, leading to dysmyelination in the PNS. Patient-derived models to study such myelination defects are lacking as the in vitro generation of human myelinating Schwann cells has proved to be particularly challenging. Here, we present an induced pluripotent stem cell-derived organoid culture, containing various cell types of the PNS, including myelinating human Schwann cells, which mimics the human PNS. Single-cell analysis confirmed the PNS-like cellular composition and provides insight into the developmental trajectory. We used this organoid model to study disease signatures of CMT1A, revealing early ultrastructural myelin alterations, including increased myelin periodic line distance and hypermyelination of small axons. Furthermore, we observed the presence of onion-bulb-like formations in a later developmental stage. These hallmarks were not present in the CMT1A-corrected isogenic line or in a CMT2A iPSC line, supporting the notion that these alterations are specific to CMT1A. Downregulation of PMP22 expression using short-hairpin RNAs or a combinatorial drug consisting of baclofen, naltrexone hydrochloride and D-sorbitol was able to ameliorate the myelin defects in CMT1A-organoids. In summary, this self-organizing organoid model can capture biologically meaningful features of the disease and capture the physiological complexity, forms an excellent model for studying demyelinating diseases and supports the therapeutic approach of reducing PMP22 expression.<\/jats:p>","DOI":"10.1093\/brain\/awac475","type":"journal-article","created":{"date-parts":[[2022,12,13]],"date-time":"2022-12-13T16:33:36Z","timestamp":1670949216000},"page":"2885-2896","source":"Crossref","is-referenced-by-count":37,"title":["Downregulation of PMP22 ameliorates myelin defects in iPSC-derived human organoid cultures of CMT1A"],"prefix":"10.1093","volume":"146","author":[{"given":"Jonas","family":"Van Lent","sequence":"first","affiliation":[{"name":"Peripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Laboratory of Neuromuscular Pathology, Institute Born Bunge, and Translational Neurosciences, Faculty of Medicine, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leen","family":"Vendredy","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Laboratory of Neuromuscular Pathology, Institute Born Bunge, and Translational Neurosciences, Faculty of Medicine, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9430-917X","authenticated-orcid":false,"given":"Elias","family":"Adriaenssens","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Laboratory of Neuromuscular Pathology, Institute Born Bunge, and Translational Neurosciences, Faculty of Medicine, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tatiana","family":"Da Silva Authier","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Laboratory of Neuromuscular Pathology, Institute Born Bunge, and Translational Neurosciences, Faculty of Medicine, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2784-2470","authenticated-orcid":false,"given":"Bob","family":"Asselbergh","sequence":"additional","affiliation":[{"name":"Neuromics Support Facility, VIB Center for Molecular Neurology, VIB , Antwerp 2610 , Belgium"},{"name":"Neuromics Support Facility, Department of Biomedical Sciences, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marcus","family":"Kaji","sequence":"additional","affiliation":[{"name":"Applied & Translational Neurogenomics Group, VIB Center for Molecular Neurology, VIB, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2878-1147","authenticated-orcid":false,"given":"Sarah","family":"Weckhuysen","sequence":"additional","affiliation":[{"name":"Applied & Translational Neurogenomics Group, VIB Center for Molecular Neurology, VIB, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Department of Neurology, Antwerp University Hospital , Antwerp 2610 , Belgium"},{"name":"Translational Neurosciences, Faculty of Medicine and Health Science, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0104-4067","authenticated-orcid":false,"given":"Ludo","family":"Van Den Bosch","sequence":"additional","affiliation":[{"name":"Department of Neurosciences, Experimental Neurology, Leuven Brain Institute, KU Leuven\u2014University of Leuven , Leuven 3000 , Belgium"},{"name":"VIB-Center for Brain & Disease Research, Laboratory of Neurobiology , Leuven 3000 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8295-5385","authenticated-orcid":false,"given":"Jonathan","family":"Baets","sequence":"additional","affiliation":[{"name":"Laboratory of Neuromuscular Pathology, Institute Born Bunge, and Translational Neurosciences, Faculty of Medicine, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Neuromuscular Reference Centre, Department of Neurology, Antwerp University Hospital , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2162-0933","authenticated-orcid":false,"given":"Vincent","family":"Timmerman","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp , Antwerp 2610 , Belgium"},{"name":"Laboratory of Neuromuscular Pathology, Institute Born Bunge, and Translational Neurosciences, Faculty of Medicine, University of Antwerp , Antwerp 2610 , Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2022,12,13]]},"reference":[{"key":"2023070312544848200_awac475-B1","doi-asserted-by":"crossref","first-page":"597","DOI":"10.1016\/j.ncl.2013.01.009","article-title":"Inherited peripheral neuropathies","volume":"31","author":"Saporta","year":"2013","journal-title":"Neurol 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Expression of peripheral myelin protein 22 in Charcot-Marie-Tooth disease type 1A and hereditary neuropathy with liability to pressure palsies nerve biopsies","volume":"49","author":"Gabriel","year":"1997","journal-title":"Neurology"},{"key":"2023070312544848200_awac475-B34","doi-asserted-by":"crossref","first-page":"121","DOI":"10.1038\/s42003-020-0854-z","article-title":"Upregulation of large myelin protein zero leads to Charcot-Marie-Tooth disease-like neuropathy in mice","volume":"3","author":"Otani","year":"2020","journal-title":"Commun Biol"},{"key":"2023070312544848200_awac475-B35","doi-asserted-by":"crossref","first-page":"389","DOI":"10.1016\/j.neuron.2018.10.007","article-title":"Building models of brain disorders with three-dimensional organoids","volume":"100","author":"Amin","year":"2018","journal-title":"Neuron"},{"key":"2023070312544848200_awac475-B36","doi-asserted-by":"crossref","first-page":"373","DOI":"10.1038\/nature12517","article-title":"Cerebral organoids model human brain development and microcephaly","volume":"501","author":"Lancaster","year":"2013","journal-title":"Nature"},{"key":"2023070312544848200_awac475-B37","first-page":"429","article-title":"Mycobacterium leprae infection of human Schwann cells depends on selective host kinases and pathogen-modulated endocytic pathways","volume":"238","author":"Alves","year":"2004","journal-title":"FEMS Microbiol Lett"},{"key":"2023070312544848200_awac475-B38","doi-asserted-by":"crossref","first-page":"1913","DOI":"10.1016\/j.cell.2020.11.017","article-title":"Generation of functional human 3D cortico-motor assembloids","volume":"183","author":"Andersen","year":"2020","journal-title":"Cell"},{"key":"2023070312544848200_awac475-B39","doi-asserted-by":"crossref","first-page":"176","DOI":"10.1186\/1742-2094-9-176","article-title":"Acute injury in the peripheral nervous system triggers an alternative macrophage response","volume":"9","author":"Ydens","year":"2012","journal-title":"J Neuroinflammation"},{"key":"2023070312544848200_awac475-B40","doi-asserted-by":"crossref","first-page":"676","DOI":"10.1038\/s41593-020-0618-6","article-title":"Profiling peripheral nerve macrophages reveals two macrophage subsets with distinct localization, transcriptome and response to injury","volume":"23","author":"Ydens","year":"2020","journal-title":"Nat Neurosci"},{"issue":"Pt 11","key":"2023070312544848200_awac475-B41","doi-asserted-by":"crossref","first-page":"3193","DOI":"10.1093\/brain\/awv240","article-title":"Targeting the colony stimulating factor 1 receptor alleviates two forms of Charcot-Marie-Tooth disease in mice","volume":"138","author":"Klein","year":"2015","journal-title":"Brain"},{"key":"2023070312544848200_awac475-B42","doi-asserted-by":"crossref","first-page":"1100","DOI":"10.1002\/glia.24158","article-title":"Early targeting of endoneurial macrophages alleviates the neuropathy and affects abnormal Schwann cell differentiation in a mouse model of Charcot-Marie-Tooth 1A","volume":"70","author":"Klein","year":"2022","journal-title":"Glia"}],"container-title":["Brain"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/brain\/advance-article-pdf\/doi\/10.1093\/brain\/awac475\/49483564\/awac475.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/brain\/article-pdf\/146\/7\/2885\/50784844\/awac475.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/brain\/article-pdf\/146\/7\/2885\/50784844\/awac475.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,7,3]],"date-time":"2023-07-03T16:21:49Z","timestamp":1688401309000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/brain\/article\/146\/7\/2885\/6895197"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,13]]},"references-count":42,"journal-issue":{"issue":"7","published-online":{"date-parts":[[2022,12,13]]},"published-print":{"date-parts":[[2023,7,3]]}},"URL":"https:\/\/doi.org\/10.1093\/brain\/awac475","relation":{},"ISSN":["0006-8950","1460-2156"],"issn-type":[{"value":"0006-8950","type":"print"},{"value":"1460-2156","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2023,7]]},"published":{"date-parts":[[2022,12,13]]}}}