{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T12:40:57Z","timestamp":1753879257523,"version":"3.41.2"},"reference-count":12,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2025,5,26]],"date-time":"2025-05-26T00:00:00Z","timestamp":1748217600000},"content-version":"vor","delay-in-days":25,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["JP19K08794"],"award-info":[{"award-number":["JP19K08794"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pigment Cell Melanoma Res"],"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:p>Mowat\u2013Wilson syndrome (MOWS) is a congenital disease characterized by intellectual disability, delayed motor development, characteristic facial features, epilepsy, and a wide spectrum of neurocristopathies. MOWS is caused by de novo heterozygous loss\u2010of\u2010function mutations or deletions in the zinc finger E\u2010box\u2010binding homeobox2 (<jats:italic>ZEB2<\/jats:italic>) gene, which is a multifunctional regulator of neuronal development and cancer progression\/metastasis through epithelial\u2010to\u2010mesenchymal transition. We recognized that patients with MOWS have brown to red hair. In the present study, we report that hair from patients with MOWS has reduced eumelanin and elevated pheomelanin contents, resulting in an increased pheomelanin\u2010to\u2010eumelanin ratio. Furthermore, <jats:italic>ZEB2<\/jats:italic>\u2010mutated human epidermal melanocytes show a predominance of pheomelanin biosynthesis over eumelanin and decreased expression of <jats:italic>SLC45A2<\/jats:italic>, the gene responsible for oculocutaneous albinism 4. Our results suggest that ZEB2 plays a role in mixed melanogenesis by regulating the melanosomal ion transporter gene, <jats:italic>SLC45A2<\/jats:italic>.<\/jats:p>","DOI":"10.1111\/pcmr.70028","type":"journal-article","created":{"date-parts":[[2025,5,26]],"date-time":"2025-05-26T08:25:30Z","timestamp":1748247930000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Alteration of Hair Melanin in Patients With Mowat\u2013Wilson Syndrome: The Role of the <i>ZEB2<\/i> Gene in Regulating Melanogenesis Through <i>SLC45A2<\/i>"],"prefix":"10.1111","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0009-0006-8605-5142","authenticated-orcid":false,"given":"Mayuko","family":"Yamamoto","sequence":"first","affiliation":[{"name":"Department of Dermatology Kochi Medical School, Kochi University  Nankoku 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