{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T09:27:29Z","timestamp":1786958849255,"version":"build-2736575974"},"reference-count":60,"publisher":"MDPI AG","issue":"16","license":[{"start":{"date-parts":[[2026,8,16]],"date-time":"2026-08-16T00:00:00Z","timestamp":1786838400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Research Funds of the Center for Advanced Interdisciplinary Science and Biomedicine of IHM","award":["QYZD20220002"],"award-info":[{"award-number":["QYZD20220002"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Biology"],"abstract":"<jats:p>Wolfram syndrome (WS) is a neurodegenerative disorder caused by mutations in the endoplasmic reticulum (ER) transmembrane protein WFS1. Mutations in WFS1 lead to ER stress and dysregulated calcium signaling, resulting in progressive neurological dysfunction. Myelination is a core pathological process in various central nervous system (CNS) diseases. However, the role of WFS1 in oligodendrocyte development and myelination remains unknown. Here, using CRISPR-Cas9-generated wfs1a\/wfs1b double-knockout (wfs1\u2212\/\u2212) zebrafish, we demonstrated that wfs1 deficiency significantly delayed the dorsal distribution of oligodendrocyte precursor cells (OPCs) along Mauthner axons. Furthermore, wfs1 mutation was associated with early hypomyelination of axons at 5 dpf, with partial recovery observed by 8 dpf. Mechanistically, combined transcriptomic and pharmacological analysis revealed that wfs1 mutation led to the suppression of the steroid biosynthesis pathway through downregulation of hmgcs1, which encodes the upstream condensation enzyme of the mevalonate pathway and supplies substrate for the downstream rate-limiting enzyme HMGCR, thereby potentially reducing isoprenoid precursor availability. Pharmacological supplementation with geranylgeraniol (GGOH) specifically rescued dorsal distribution defects, supporting involvement of the isoprenylation pathway, although direct regulation of hmgcs1 by wfs1 and complete rescue of myelin structure require further mechanistic validation. Taken together, our study supports a novel wfs1-hmgcs1 axis, which may regulate dorsal distribution and myelination through the isoprenylation pathway; notably, classical ER stress markers were also concurrently upregulated in wfs1\u2212\/\u2212 larvae, although whether the isoprenylation deficiency and ER stress act independently or synergistically remains to be determined. These findings provide new insights into WS-associated early hypomyelination and suggest hmgcs1 as a potential therapeutic target for myelin defects caused by WS.<\/jats:p>","DOI":"10.3390\/biology15161402","type":"journal-article","created":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T08:05:41Z","timestamp":1786953941000},"page":"1402","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Deletion of wfs1 Impairs Oligodendrocyte Precursor Cells Dorsal Distribution and Myelination Through the wfs1-hmgcs1 Axis in Zebrafish"],"prefix":"10.3390","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0009-0005-9058-8104","authenticated-orcid":false,"given":"Xiahui","family":"Tang","sequence":"first","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-1861-6182","authenticated-orcid":false,"given":"Ziang","family":"Zhao","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kunlun","family":"Yao","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-7234-5618","authenticated-orcid":false,"given":"Dinggang","family":"Fan","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-6148-2106","authenticated-orcid":false,"given":"Keqiang","family":"Li","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Junhui","family":"Zhou","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6314-5782","authenticated-orcid":false,"given":"Zongyi","family":"Wang","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"},{"name":"Department of Psychiatry and Behavioral Sciences, Stanford University School of Medicine, Stanford, CA 94305, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8627-5272","authenticated-orcid":false,"given":"Bing","family":"Hu","sequence":"additional","affiliation":[{"name":"Center for Advanced Interdisciplinary Science and Biomedicine of Institute of Health and Medicine, Division of Life Sciences and Medicine, University of Science and Technology of China, Hefei 230026, China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2026,8,16]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"10220","DOI":"10.1038\/s41598-017-09392-x","article-title":"Wfs1- Deficient Rats Develop Primary Symptoms of Wolfram Syndrome: Insulin-Dependent Diabetes, Optic Nerve Atrophy and Medullary Degeneration","volume":"7","author":"Plaas","year":"2017","journal-title":"Sci. Rep."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"21167","DOI":"10.1038\/srep21167","article-title":"Washington University Wolfram Syndrome Research Study Group Neuroimaging Evidence of Deficient Axon Myelination in Wolfram Syndrome","volume":"6","author":"Lugar","year":"2016","journal-title":"Sci. Rep."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1007\/s00401-009-0546-8","article-title":"Wolfram Syndrome: A Clinicopathologic Correlation","volume":"118","author":"Hilson","year":"2009","journal-title":"Acta Neuropathol."},{"key":"ref_4","doi-asserted-by":"crossref","unstructured":"Rigoli, L., Caruso, V., Salzano, G., and Lombardo, F. (2022). Wolfram Syndrome 1: From Genetics to Therapy. Int. J. Environ. Res. Public. Health, 19.","DOI":"10.3390\/ijerph19063225"},{"key":"ref_5","doi-asserted-by":"crossref","unstructured":"Serbis, A., Rallis, D., Giapros, V., Galli-Tsinopoulou, A., and Siomou, E. (2023). Wolfram Syndrome 1: A Pediatrician\u2019s and Pediatric Endocrinologist\u2019s Perspective. Int. J. Mol. Sci., 24.","DOI":"10.3390\/ijms24043690"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"744","DOI":"10.1172\/JCI39678","article-title":"Wolfram Syndrome 1 Gene Negatively Regulates ER Stress Signaling in Rodent and Human Cells","volume":"120","author":"Fonseca","year":"2010","journal-title":"J. Clin. Investig."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"849","DOI":"10.1038\/s41374-020-0408-5","article-title":"Wolfram Syndrome 1 Gene Regulates Pathways Maintaining Beta-Cell Health and Survival","volume":"100","author":"Abreu","year":"2020","journal-title":"Lab. Investig. J. Tech. Methods Pathol."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"2055","DOI":"10.1080\/15548627.2024.2341588","article-title":"The Wolfram-like Variant WFS1E864K Destabilizes MAM and Compromises Autophagy and Mitophagy in Human and Mice","volume":"20","author":"Patergnani","year":"2024","journal-title":"Autophagy"},{"key":"ref_9","doi-asserted-by":"crossref","unstructured":"K\u00f5ks, S. (2023). Genomics of Wolfram Syndrome 1 (WFS1). Biomolecules, 13.","DOI":"10.3390\/biom13091346"},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"921","DOI":"10.1038\/pr.2018.17","article-title":"Genetic and Clinical Aspects of Wolfram Syndrome 1, a Severe Neurodegenerative Disease","volume":"83","author":"Rigoli","year":"2018","journal-title":"Pediatr. Res."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1007\/s11892-015-0702-6","article-title":"Wolfram Syndrome: Diagnosis, Management, and Treatment","volume":"16","author":"Urano","year":"2016","journal-title":"Curr. Diab. Rep."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"279","DOI":"10.1186\/s13023-019-1260-9","article-title":"Developmental Hypomyelination in Wolfram Syndrome: New Insights from Neuroimaging and Gene Expression Analyses","volume":"14","author":"Samara","year":"2019","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1038\/s41586-022-05534-y","article-title":"Microglia Regulate Central Nervous System Myelin Growth and Integrity","volume":"613","author":"McNamara","year":"2023","journal-title":"Nature"},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"102194","DOI":"10.1016\/j.coph.2022.102194","article-title":"Breaking the Barriers to Remyelination in Multiple Sclerosis","volume":"63","author":"Gharagozloo","year":"2022","journal-title":"Curr. Opin. Pharmacol."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"93","DOI":"10.1016\/j.conb.2016.04.013","article-title":"The Cell Biology of CNS Myelination","volume":"39","author":"Hughes","year":"2016","journal-title":"Curr. Opin. Neurobiol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"a020453","DOI":"10.1101\/cshperspect.a020453","article-title":"Oligodendrocyte Development and Plasticity","volume":"8","author":"Bergles","year":"2015","journal-title":"Cold Spring Harb. Perspect. Biol."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"363","DOI":"10.1038\/s41593-019-0581-2","article-title":"Functionally Distinct Subgroups of Oligodendrocyte Precursor Cells Integrate Neural Activity and Execute Myelin Formation","volume":"23","author":"Marisca","year":"2020","journal-title":"Nat. Neurosci."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"628","DOI":"10.1016\/j.tins.2023.05.007","article-title":"Emerging Roles of Oligodendrocyte Precursor Cells in Neural Circuit Development and Remodeling","volume":"46","author":"Buchanan","year":"2023","journal-title":"Trends Neurosci."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"350","DOI":"10.1038\/s41419-024-06737-z","article-title":"Seipin Deficiency-Induced Lipid Dysregulation Leads to Hypomyelination-Associated Cognitive Deficits via Compromising Oligodendrocyte Precursor Cell Differentiation","volume":"15","author":"Cui","year":"2024","journal-title":"Cell Death Dis."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"110423","DOI":"10.1016\/j.celrep.2022.110423","article-title":"Cholesterol Biosynthesis Defines Oligodendrocyte Precursor Heterogeneity between Brain and Spinal Cord","volume":"38","author":"Khandker","year":"2022","journal-title":"Cell Rep."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1007\/s11481-025-10236-z","article-title":"Emerging Role of Oligodendrocytes Malfunction in the Progression of Alzheimer\u2019s Disease","volume":"20","author":"Tylek","year":"2025","journal-title":"J. Neuroimmune Pharmacol. Off. J. Soc. NeuroImmune Pharmacol."},{"key":"ref_22","first-page":"e00540","article-title":"Astrocyte and Oligodendrocyte Pathology in Alzheimer\u2019s Disease","volume":"22","author":"Ziar","year":"2025","journal-title":"Neurother. J. Am. Soc. Exp. Neurother."},{"key":"ref_23","doi-asserted-by":"crossref","unstructured":"Chacon-De-La-Rocha, I., Fryatt, G., Rivera, A.D., Verkhratsky, A., Raineteau, O., Gomez-Nicola, D., and Butt, A.M. (2020). Accelerated Dystrophy and Decay of Oligodendrocyte Precursor Cells in the APP\/PS1 Model of Alzheimer\u2019s-Like Pathology. Front. Cell. Neurosci., 14.","DOI":"10.3389\/fncel.2020.575082"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"1452","DOI":"10.1038\/ng.2802","article-title":"Meta-Analysis of 74,046 Individuals Identifies 11 New Susceptibility Loci for Alzheimer\u2019s Disease","volume":"45","author":"Lambert","year":"2013","journal-title":"Nat. Genet."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"1501","DOI":"10.4103\/1673-5374.361540","article-title":"New Unexpected Role for Wolfram Syndrome Protein WFS1: A Novel Therapeutic Target for Alzheimer\u2019s Disease?","volume":"18","author":"Chen","year":"2023","journal-title":"Neural Regen. Res."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"2711","DOI":"10.1093\/hmg\/ddac065","article-title":"Morphological, Behavioral and Cellular Analyses Revealed Different Phenotypes in Wolfram Syndrome Wfs1a and Wfs1b Zebrafish Mutant Lines","volume":"31","author":"Crouzier","year":"2022","journal-title":"Hum. Mol. Genet."},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"20491","DOI":"10.1038\/s41598-021-99781-0","article-title":"A Mutant Wfs1 Zebrafish Model of Wolfram Syndrome Manifesting Visual Dysfunction and Developmental Delay","volume":"11","author":"Cairns","year":"2021","journal-title":"Sci. Rep."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"387","DOI":"10.1038\/s41419-023-05912-y","article-title":"Wfs1E864K Knock-in Mice Illuminate the Fundamental Role of Wfs1 in Endocochlear Potential Production","volume":"14","author":"Richard","year":"2023","journal-title":"Cell Death Dis."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"140","DOI":"10.1186\/s40478-024-01851-7","article-title":"A Deep Phenotyping Study in Mouse and iPSC Models to Understand the Role of Oligodendroglia in Optic Neuropathy in Wolfram Syndrome","volume":"12","author":"Ahuja","year":"2024","journal-title":"Acta Neuropathol. Commun."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"3402","DOI":"10.1523\/JNEUROSCI.4587-13.2014","article-title":"Mutation of 3-Hydroxy-3-Methylglutaryl CoA Synthase I Reveals Requirements for Isoprenoid and Cholesterol Synthesis in Oligodendrocyte Migration Arrest, Axon Wrapping, and Myelin Gene Expression","volume":"34","author":"Mathews","year":"2014","journal-title":"J. Neurosci. Off. J. Soc. Neurosci."},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"1147","DOI":"10.1002\/glia.24336","article-title":"Emerging Concepts in Oligodendrocyte and Myelin Formation, Inputs from the Zebrafish Model","volume":"71","author":"Masson","year":"2023","journal-title":"Glia"},{"key":"ref_32","doi-asserted-by":"crossref","unstructured":"Yang, L.-Q., Chen, M., Zhang, J.-L., Ren, D.-L., and Hu, B. (2018). Hypoxia Delays Oligodendrocyte Progenitor Cell Migration and Myelin Formation by Suppressing Bmp2b Signaling in Larval Zebrafish. Front. Cell. Neurosci., 12.","DOI":"10.3389\/fncel.2018.00348"},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"1506","DOI":"10.1038\/nn1803","article-title":"In Vivo Time-Lapse Imaging Shows Dynamic Oligodendrocyte Progenitor Behavior during Zebrafish Development","volume":"9","author":"Kirby","year":"2006","journal-title":"Nat. Neurosci."},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"333","DOI":"10.1002\/glia.22897","article-title":"Insights into Mechanisms of Central Nervous System Myelination Using Zebrafish","volume":"64","author":"Czopka","year":"2016","journal-title":"Glia"},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"962","DOI":"10.1038\/cr.2013.62","article-title":"Developmental Pruning of Early-Stage Myelin Segments during CNS Myelination in Vivo","volume":"23","author":"Liu","year":"2013","journal-title":"Cell Res."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"1790","DOI":"10.1038\/s41467-024-45908-6","article-title":"Importin 13-Dependent Axon Diameter Growth Regulates Conduction Speeds along Myelinated CNS Axons","volume":"15","author":"Bin","year":"2024","journal-title":"Nat. Commun."},{"key":"ref_37","first-page":"4443","article-title":"Individual Axons Regulate the Myelinating Potential of Single Oligodendrocytes in Vivo","volume":"138","author":"Almeida","year":"2011","journal-title":"Dev. Camb. Engl."},{"key":"ref_38","doi-asserted-by":"crossref","unstructured":"Shi, L., Wang, Z., Li, Y., Song, Z., Yin, W., and Hu, B. (2023). Deletion of the Chd7 Hinders Oligodendrocyte Progenitor Cell Development and Myelination in Zebrafish. Int. J. Mol. Sci., 24.","DOI":"10.3390\/ijms241713535"},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1023\/B:MICS.0000006847.09037.3a","article-title":"Neural Cell Fate Analysis in Zebrafish Using Olig2 BAC Transgenics","volume":"25","author":"Shin","year":"2003","journal-title":"Methods Cell Sci. Off. J. Soc. Vitr. Biol."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"e66080","DOI":"10.7554\/eLife.66080","article-title":"In Vivo Fluorescence Lifetime Imaging of Macrophage Intracellular Metabolism during Wound Responses in Zebrafish","volume":"11","author":"Miskolci","year":"2022","journal-title":"eLife"},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"W242","DOI":"10.1093\/nar\/gky354","article-title":"CRISPOR: Intuitive Guide Selection for CRISPR\/Cas9 Genome Editing Experiments and Screens","volume":"46","author":"Concordet","year":"2018","journal-title":"Nucleic Acids Res."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"184","DOI":"10.1186\/s40478-022-01484-8","article-title":"Wolfram Syndrome 1b Mutation Suppresses Mauthner-Cell Axon Regeneration via ER Stress Signal Pathway","volume":"10","author":"Wang","year":"2022","journal-title":"Acta Neuropathol. Commun."},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"515","DOI":"10.1002\/cne.25440","article-title":"Oligodendrocyte Origin and Development in the Zebrafish Visual System","volume":"531","author":"Velasco","year":"2023","journal-title":"J. Comp. Neurol."},{"key":"ref_44","doi-asserted-by":"crossref","unstructured":"Collins, S.C., Vancollie, V.E., Mikhaleva, A., Wagner, C., Balz, R., Lelliott, C.J., and Yalcin, B. (2022). Characterization of Two Mouse Chd7 Heterozygous Loss-of-Function Models Shows Dysgenesis of the Corpus Callosum and Previously Unreported Features of CHARGE Syndrome. Int. J. Mol. Sci., 23.","DOI":"10.3390\/ijms231911509"},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"2445","DOI":"10.1016\/j.cub.2018.06.016","article-title":"Forebrain Control of Behaviorally Driven Social Orienting in Zebrafish","volume":"28","author":"Stednitz","year":"2018","journal-title":"Curr. Biol. CB"},{"key":"ref_46","first-page":"33","article-title":"Larval Rearing Methods for Small-Scale Production of Healthy Zebrafish","volume":"2019","author":"Norton","year":"2019","journal-title":"East. Biol."},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"3336","DOI":"10.1002\/dvdy.22455","article-title":"Expression of the Transcription Factor Olig2 in Proliferating Cells in the Adult Zebrafish Telencephalon","volume":"239","author":"Schmidt","year":"2010","journal-title":"Dev. Dyn."},{"key":"ref_48","doi-asserted-by":"crossref","unstructured":"Kroehne, V., Tsata, V., Marrone, L., Froeb, C., Reinhardt, S., Gompf, A., Dahl, A., Sterneckert, J., and Reimer, M.M. (2017). Primary Spinal OPC Culture System from Adult Zebrafish to Study Oligodendrocyte Differentiation In Vitro. Front. Cell. Neurosci., 11.","DOI":"10.3389\/fncel.2017.00284"},{"key":"ref_49","doi-asserted-by":"crossref","first-page":"dev193946","DOI":"10.1242\/dev.193946","article-title":"Reactive Oligodendrocyte Progenitor Cells (Re-)Myelinate the Regenerating Zebrafish Spinal Cord","volume":"147","author":"Tsata","year":"2020","journal-title":"Development"},{"key":"ref_50","doi-asserted-by":"crossref","first-page":"177","DOI":"10.1002\/glia.22755","article-title":"Zebrafish as a Model to Investigate CNS Myelination","volume":"63","author":"Preston","year":"2015","journal-title":"Glia"},{"key":"ref_51","doi-asserted-by":"crossref","first-page":"e201910213","DOI":"10.1083\/jcb.201910213","article-title":"TDP-43 Mediates SREBF2-Regulated Gene Expression Required for Oligodendrocyte Myelination","volume":"220","author":"Ho","year":"2021","journal-title":"J. Cell Biol."},{"key":"ref_52","doi-asserted-by":"crossref","first-page":"985","DOI":"10.1002\/dneu.22856","article-title":"Mechanisms of Oligodendrocyte Progenitor Developmental Migration","volume":"81","author":"Xia","year":"2021","journal-title":"Dev. Neurobiol."},{"key":"ref_53","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1016\/j.ceb.2021.05.003","article-title":"Oligodendrocyte Progenitor Cell Fate and Function in Development and Disease","volume":"73","author":"Clayton","year":"2021","journal-title":"Curr. Opin. Cell Biol."},{"key":"ref_54","doi-asserted-by":"crossref","first-page":"6996","DOI":"10.1038\/s41467-021-27344-y","article-title":"WFS1 Functions in ER Export of Vesicular Cargo Proteins in Pancreatic \u03b2-Cells","volume":"12","author":"Wang","year":"2021","journal-title":"Nat. Commun."},{"key":"ref_55","doi-asserted-by":"crossref","unstructured":"Waszczykowska, A., Zmys\u0142owska, A., Braun, M., Ivask, M., Koks, S., Jurowski, P., and M\u0142ynarski, W. (2020). Multiple Retinal Anomalies in Wfs1-Deficient Mice. Diagn. Basel Switz., 10.","DOI":"10.3390\/diagnostics10090607"},{"key":"ref_56","doi-asserted-by":"crossref","first-page":"379","DOI":"10.1038\/nn.2273","article-title":"Endoplasmic Reticulum Stress in Disorders of Myelinating Cells","volume":"12","author":"Lin","year":"2009","journal-title":"Nat. Neurosci."},{"key":"ref_57","doi-asserted-by":"crossref","first-page":"648","DOI":"10.1016\/j.stemcr.2014.03.007","article-title":"Involvement of ER Stress in Dysmyelination of Pelizaeus-Merzbacher Disease with PLP1 Missense Mutations Shown by iPSC-Derived Oligodendrocytes","volume":"2","author":"Okada","year":"2014","journal-title":"Stem Cell Rep."},{"key":"ref_58","doi-asserted-by":"crossref","first-page":"3113","DOI":"10.1093\/brain\/awae158","article-title":"PMP22 Duplication Dysregulates Lipid Homeostasis and Plasma Membrane Organization in Developing Human Schwann Cells","volume":"147","author":"Prior","year":"2024","journal-title":"Brain J. Neurol."},{"key":"ref_59","doi-asserted-by":"crossref","first-page":"215761","DOI":"10.1016\/j.canlet.2022.215761","article-title":"The Mevalonate Pathway in Breast Cancer Biology","volume":"542","author":"Riffel","year":"2022","journal-title":"Cancer Lett."},{"key":"ref_60","doi-asserted-by":"crossref","first-page":"10130","DOI":"10.1073\/pnas.1821306116","article-title":"Gene Expression in Oligodendrocytes during Remyelination Reveals Cholesterol Homeostasis as a Therapeutic Target in Multiple Sclerosis","volume":"116","author":"Voskuhl","year":"2019","journal-title":"Proc. Natl. Acad. Sci. USA"}],"container-title":["Biology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2079-7737\/15\/16\/1402\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T08:52:41Z","timestamp":1786956761000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2079-7737\/15\/16\/1402"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,8,16]]},"references-count":60,"journal-issue":{"issue":"16","published-online":{"date-parts":[[2026,8]]}},"alternative-id":["biology15161402"],"URL":"https:\/\/doi.org\/10.3390\/biology15161402","relation":{},"ISSN":["2079-7737"],"issn-type":[{"value":"2079-7737","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,8,16]]}}}