{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,23]],"date-time":"2026-04-23T04:31:54Z","timestamp":1776918714352,"version":"3.51.2"},"reference-count":26,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2023,3,15]],"date-time":"2023-03-15T00:00:00Z","timestamp":1678838400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100003716","name":"Korea Basic Science Institute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003716","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003725","name":"National Research Foundation of Korea","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003725","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2023,6]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background and purpose<\/jats:title><jats:p>Elevated plasma concentrations of neural cell adhesion molecule 1 (NCAM1) and p75 neurotrophin receptor (p75) in patients with peripheral neuropathy have been reported. This study aimed to determine the specificity of plasma concentration elevation of either NCAM1 or p75 in a subtype of Charcot\u2013Marie\u2013Tooth disease (CMT) and its correlation with pathologic nerve status and disease severity.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Blood samples were collected from 138 patients with inherited peripheral neuropathy and 51 healthy controls. Disease severity was measured using Charcot\u2013Marie\u2013Tooth Neuropathy Score version 2 (CMTNSv2), and plasma concentrations of NCAM1 and p75 were analyzed by enzyme\u2010linked immunosorbent assay. Eight sural nerves from CMT patients were examined to determine the relation of histopathology and plasma NCAM1 levels.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Plasma concentration of NCAM1, but not p75, was specifically increased in demyelinating subtypes of CMT (median\u00a0=\u00a07100\u2009pg\/mL, <jats:italic>p\u2009&lt;<\/jats:italic>\u00a00.001), including CMT1A, but not in axonal subtype (5964\u2009pg\/mL, <jats:italic>p\u2009&gt;<\/jats:italic>\u00a00.05), compared to the control (3859\u2009pg\/mL). CMT1A patients with mild or moderate severity (CMTNSv2 &lt;\u200920) showed higher levels of plasma NCAM1 than healthy controls. Immunofluorescent NCAM1 staining for the sural nerves of CMT patients showed that NCAM1\u2010positive onion bulb cells and possible demyelinating Schwann cells might be associated with the specific increase of plasma NCAM1 in demyelinating CMT.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>The plasma NCAM1 levels in demyelinating CMT might be a surrogate biomarker reflecting pathological Schwann cell status and disease progression.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ene.15763","type":"journal-article","created":{"date-parts":[[2023,3,1]],"date-time":"2023-03-01T14:40:02Z","timestamp":1677681602000},"page":"1745-1754","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Neural cell adhesion molecule 1 is a cellular target engaged plasma biomarker in demyelinating <scp>Charcot\u2013Marie\u2013Tooth<\/scp> disease"],"prefix":"10.1111","volume":"30","author":[{"given":"Young Hee","family":"Kim","sequence":"first","affiliation":[{"name":"Peripheral Neuropathy Research Center Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Byeol\u2010A","family":"Yoon","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Center Dong\u2010A University College of Medicine  Busan Republic of Korea"},{"name":"Department of Neurology Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Young Rae","family":"Jo","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Center Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Soo Hyun","family":"Nam","sequence":"additional","affiliation":[{"name":"Stem Cell and Regenerative Medicine Institute Samsung Medical Center  Seoul Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nam Hee","family":"Kim","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kyoung Hee","family":"Kim","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jong Kuk","family":"Kim","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Center Dong\u2010A University College of Medicine  Busan Republic of Korea"},{"name":"Department of Neurology Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Byung\u2010Ok","family":"Choi","sequence":"additional","affiliation":[{"name":"Stem Cell and Regenerative Medicine Institute Samsung Medical Center  Seoul Republic of Korea"},{"name":"Department of Neurology, Samsung Medical Center Sungkyunkwan University School of Medicine  Seoul Republic of Korea"},{"name":"Department of Health Sciences and Technology The Samsung Advanced Institute for Health Sciences &amp; Technology, Sungkyunkwan University  Seoul Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0913-1221","authenticated-orcid":false,"given":"Hwan Tae","family":"Park","sequence":"additional","affiliation":[{"name":"Peripheral Neuropathy Research Center Dong\u2010A University College of Medicine  Busan Republic of Korea"},{"name":"Department of Molecular Neuroscience Dong\u2010A University College of Medicine  Busan Republic of Korea"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2023,3,15]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2014-308826"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.brainres.2019.146625"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000006214"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/ene.14689"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000004932"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2014-308826"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/7.3.449"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awr322"},{"issue":"7","key":"e_1_2_8_10_1","first-page":"1516","article-title":"Neurological dysfunction and axonal degeneration in Charcot\u2013Marie\u2013tooth disease type 1A","volume":"123","author":"Krajewski KM","year":"2000","journal-title":"Brain Res"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/acn3.50828"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.25480"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/glia.23509"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1007\/s004010050712"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awac055"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/glia.20761"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/119.5.1461"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1529-8027.2011.00350.x"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2990.1993.tb00478.x"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00318579"},{"issue":"1","key":"e_1_2_8_21_1","first-page":"3025","article-title":"Targeting myelin lipid metabolism as a potential therapeutic strategy in a model of CMT1A neuropathy. 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