{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T18:33:23Z","timestamp":1777919603549,"version":"3.51.4"},"reference-count":29,"publisher":"SAGE Publications","issue":"2","license":[{"start":{"date-parts":[[2025,10,9]],"date-time":"2025-10-09T00:00:00Z","timestamp":1759968000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Vet Pathol"],"published-print":{"date-parts":[[2026,3]]},"abstract":"<jats:p>\n                    The vacuole formation (VF) rat is an autosomal recessive myelin mutant characterized by generalized tremor, hypomyelination, and periaxonal VF of the central nervous system. We previously identified a nonsense mutation in the\n                    <jats:italic toggle=\"yes\">DOP1 leucine zipper-like protein A<\/jats:italic>\n                    (\n                    <jats:italic toggle=\"yes\">Dop1a<\/jats:italic>\n                    , also known as\n                    <jats:italic toggle=\"yes\">Dopey1<\/jats:italic>\n                    ) gene located on rat chromosome 8. In this study, we investigated the kinetics of oligodendrocyte progenitor cells (OPCs) and oligodendrocytes by assessing the expression patterns of transcription factors that are involved in oligodendrocyte development. The number of small, round oligodendrocytes lacking distinct cellular processes, immunolabeled with anti-adenomatous polyposis coli (APC, clone CC1) antibody, was increased in the spinal cords of the VF rats, suggesting a disrupted maturation of oligodendrocytes. In addition, the terminal differentiation and maturation of OPCs into myelinating mature oligodendrocytes may be impaired and compensatory myelination largely could fail in the VF rat. Our findings also demonstrated that the DOP1A protein is expressed in OPCs as well as mature oligodendrocytes. Finally, the intracellular trafficking of\n                    <jats:italic toggle=\"yes\">myelin basic protein<\/jats:italic>\n                    (\n                    <jats:italic toggle=\"yes\">Mbp<\/jats:italic>\n                    ) mRNAs may be disrupted in oligodendrocytes of the VF rats. Our data suggested that DOP1A dysfunction causes impaired differentiation and maturation of oligodendrocyte-lineage cells, resulting in hypomyelination and an impaired compensatory myelination.\n                  <\/jats:p>","DOI":"10.1177\/03009858251379486","type":"journal-article","created":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T01:04:26Z","timestamp":1760058266000},"page":"315-324","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Impaired maturation and differentiation of oligodendrocytes in the vacuole formation myelin mutant rat"],"prefix":"10.1177","volume":"63","author":[{"ORCID":"https:\/\/orcid.org\/0009-0006-8237-260X","authenticated-orcid":false,"given":"Miyuu","family":"Tanaka","sequence":"first","affiliation":[{"name":"Osaka Metropolitan University, Izumisano, Japan"},{"name":"Kyoto University, Kyoto, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9317-8876","authenticated-orcid":false,"given":"Chiaki","family":"Morita","sequence":"additional","affiliation":[{"name":"Osaka Metropolitan University, Izumisano, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3236-6050","authenticated-orcid":false,"given":"Takeshi","family":"Izawa","sequence":"additional","affiliation":[{"name":"Osaka Metropolitan University, Izumisano, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jyoji","family":"Yamate","sequence":"additional","affiliation":[{"name":"Osaka Metropolitan University, Izumisano, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6522-2104","authenticated-orcid":false,"given":"Robin J. M.","family":"Franklin","sequence":"additional","affiliation":[{"name":"University of Cambridge, Addenbrookes Hospital, Cambridge, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Takashi","family":"Kuramoto","sequence":"additional","affiliation":[{"name":"Kyoto University, Kyoto, Japan"},{"name":"Tokyo University of Agriculture, Atsugi, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-6964-5808","authenticated-orcid":false,"given":"Tadao","family":"Serikawa","sequence":"additional","affiliation":[{"name":"Kyoto University, Kyoto, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-6577-2252","authenticated-orcid":false,"given":"Mitsuru","family":"Kuwamura","sequence":"additional","affiliation":[{"name":"Osaka Metropolitan University, Izumisano, Japan"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2025,10,9]]},"reference":[{"key":"e_1_3_3_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.febslet.2009.10.085"},{"key":"e_1_3_3_3_2","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.147.6.1123"},{"key":"e_1_3_3_4_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.118.1.283"},{"key":"e_1_3_3_5_2","doi-asserted-by":"publisher","DOI":"10.1152\/physrev.2001.81.2.871"},{"key":"e_1_3_3_6_2","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.19-17-07278.1999"},{"key":"e_1_3_3_7_2","doi-asserted-by":"publisher","DOI":"10.1038\/cdd.2008.70"},{"key":"e_1_3_3_8_2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-neuro-061010-113629"},{"key":"e_1_3_3_9_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.mcn.2004.06.015"},{"key":"e_1_3_3_10_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.129.3.681"},{"key":"e_1_3_3_11_2","doi-asserted-by":"publisher","DOI":"10.1046\/j.1471-4159.2000.0751834.x"},{"key":"e_1_3_3_12_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.122.12.4085"},{"key":"e_1_3_3_13_2","doi-asserted-by":"publisher","DOI":"10.1002\/jemt.1050"},{"key":"e_1_3_3_14_2","doi-asserted-by":"publisher","DOI":"10.1023\/A:1021667515030"},{"key":"e_1_3_3_15_2","doi-asserted-by":"publisher","DOI":"10.1023\/A:1019693421778"},{"key":"e_1_3_3_16_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuroscience.2013.11.029"},{"key":"e_1_3_3_17_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.118.2.539"},{"key":"e_1_3_3_18_2","doi-asserted-by":"publisher","DOI":"10.1538\/expanim.51.149"},{"key":"e_1_3_3_19_2","doi-asserted-by":"publisher","DOI":"10.1242\/dev.118.2.563"},{"issue":"3","key":"e_1_3_3_20_2","first-page":"261","article-title":"Insertion of a retrotransposon in Mbp disrupts mRNA splicing and myelination in a new mutant rat","volume":"281","author":"O\u2019Connor LT","year":"2009","unstructured":"O\u2019Connor LT, Goetz BD, Kwiecien JM, et al. Insertion of a retrotransposon in Mbp disrupts mRNA splicing and myelination in a new mutant rat. 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