{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T05:30:40Z","timestamp":1790400640923,"version":"4.1.0"},"reference-count":48,"publisher":"Oxford University Press (OUP)","issue":"21","license":[{"start":{"date-parts":[[2019,8,14]],"date-time":"2019-08-14T00:00:00Z","timestamp":1565740800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"name":"Wellcome Trust Postdoctoral Fellowship for Clinicians","award":["110043\/Z\/15\/Z"],"award-info":[{"award-number":["110043\/Z\/15\/Z"]}]},{"DOI":"10.13039\/501100001810","name":"Research Promotion Foundation","doi-asserted-by":"publisher","award":["CULTURE\/BR-NE\/0416\/07"],"award-info":[{"award-number":["CULTURE\/BR-NE\/0416\/07"]}],"id":[{"id":"10.13039\/501100001810","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Muscular Dystrophy Association and Charcot-Marie-Tooth Association","award":["603003"],"award-info":[{"award-number":["603003"]}]},{"name":"Muscular Dystrophy Association and Charcot-Marie-Tooth Association","award":["480030"],"award-info":[{"award-number":["480030"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,11,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>X-linked Charcot-Marie-Tooth disease (CMT1X), one of the commonest forms of inherited demyelinating neuropathy, results from GJB1 gene mutations causing loss of function of the gap junction protein connexin32 (Cx32). The aim of this study was to examine whether delayed gene replacement therapy after the onset of peripheral neuropathy can provide a therapeutic benefit in the Gjb1-null\/Cx32 knockout model of CMT1X. After delivery of the LV-Mpz.GJB1 lentiviral vector by a single lumbar intrathecal injection into 6-month-old Gjb1-null mice, we confirmed expression of Cx32 in lumbar roots and sciatic nerves correctly localized at the paranodal myelin areas. Gjb1-null mice treated with LV-Mpz.GJB1 compared with LV-Mpz.Egfp (mock) vector at the age of 6\u00a0months showed improved motor performance at 8 and 10\u00a0months. Furthermore, treated mice showed increased sciatic nerve conduction velocities, improvement of myelination and reduced inflammation in lumbar roots and peripheral nerves at 10\u00a0months of age, along with enhanced quadriceps muscle innervation. Plasma neurofilament light (NEFL) levels, a clinically relevant biomarker, were also ameliorated in fully treated mice. Intrathecal gene delivery after the onset of peripheral neuropathy offers a significant therapeutic benefit in this disease model, providing a proof of principle for treating patients with CMT1X at different ages.<\/jats:p>","DOI":"10.1093\/hmg\/ddz199","type":"journal-article","created":{"date-parts":[[2019,8,6]],"date-time":"2019-08-06T15:11:17Z","timestamp":1565104277000},"page":"3528-3542","source":"Crossref","is-referenced-by-count":40,"title":["Gene replacement therapy after neuropathy onset provides therapeutic benefit in a model of CMT1X"],"prefix":"10.1093","volume":"28","author":[{"given":"A","family":"Kagiava","sequence":"first","affiliation":[{"name":"Neuroscience Laboratory, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J","family":"Richter","sequence":"additional","affiliation":[{"name":"Department of Molecular Virology, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C","family":"Tryfonos","sequence":"additional","affiliation":[{"name":"Department of Molecular Virology, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C","family":"Karaiskos","sequence":"additional","affiliation":[{"name":"Neuroscience Laboratory, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A J","family":"Heslegrave","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"I","family":"Sargiannidou","sequence":"additional","affiliation":[{"name":"Neuroscience Laboratory, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A M","family":"Rossor","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H","family":"Zetterberg","sequence":"additional","affiliation":[{"name":"Department of Neurodegenerative Disease, UCL Institute of Neurology, London, United Kingdom"},{"name":"UK Dementia Research Institute at UCL, London, United Kingdom"},{"name":"Department of Psychiatry and Neurochemistry, Institute of Neuroscience and Physiology, the Sahlgrenska Academy at the University of Gothenburg, M\u00f6lndal, Sweden"},{"name":"Clinical Neurochemistry Laboratory, Sahlgrenska University Hospital, M\u00f6lndal, Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M M","family":"Reilly","sequence":"additional","affiliation":[{"name":"Department of Neuromuscular Diseases, UCL Queen Square Institute of Neurology, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"C","family":"Christodoulou","sequence":"additional","affiliation":[{"name":"Department of Molecular Virology, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K A","family":"Kleopa","sequence":"additional","affiliation":[{"name":"Neuroscience Laboratory, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"},{"name":"Neurology Clinics, The Cyprus Institute of Neurology and Genetics and Cyprus School of Molecular Medicine, Nicosia, Cyprus"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2019,8,14]]},"reference":[{"key":"2019122311031839100_ref1","doi-asserted-by":"crossref","first-page":"849","DOI":"10.1212\/01.wnl.0000256709.08271.4d","article-title":"CMT1X phenotypes represent loss of GJB1 gene function","volume":"68","author":"Shy","year":"2007","journal-title":"Neurology"},{"key":"2019122311031839100_ref2","doi-asserted-by":"crossref","first-page":"2030","DOI":"10.1016\/j.bbamem.2011.08.015","article-title":"Gap junctions in inherited human disorders of the central nervous system","volume":"1818","author":"Abrams","year":"2012","journal-title":"Biochim. Biophys. Acta"},{"key":"2019122311031839100_ref3","doi-asserted-by":"crossref","first-page":"107","DOI":"10.1385\/NMM:8:1-2:107","article-title":"Molecular genetics of X-linked Charcot\u2013Marie\u2013Tooth disease","volume":"8","author":"Kleopa","year":"2006","journal-title":"Neuromolecular Med."},{"key":"2019122311031839100_ref4","doi-asserted-by":"crossref","first-page":"396","DOI":"10.1212\/01.wnl.0000196479.93722.59","article-title":"Phenotypic and cellular expression of two novel connexin32 mutations causing CMT1X","volume":"66","author":"Kleopa","year":"2006","journal-title":"Neurology"},{"key":"2019122311031839100_ref5","doi-asserted-by":"crossref","first-page":"429","DOI":"10.1002\/ana.10305","article-title":"Transient central nervous system white matter abnormality in X-linked Charcot\u2013Marie\u2013Tooth disease","volume":"52","author":"Paulson","year":"2002","journal-title":"Ann. 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