{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,19]],"date-time":"2026-10-19T23:38:48Z","timestamp":1792453128759,"version":"4.5.0"},"reference-count":44,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2025,10,6]],"date-time":"2025-10-06T00:00:00Z","timestamp":1759708800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["32170973"],"award-info":[{"award-number":["32170973"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["nyaspubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Ann NY Acad Sci"],"published-print":{"date-parts":[[2025,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Lipids are essential for brain development, functioning as both structural components of cell membranes and key signaling molecules. However, the regulatory mechanisms underlying lipid metabolism during neurodevelopment remain incompletely understood. The gene disconnected interacting protein 2 homolog C (\n                    <jats:italic>DIP2C<\/jats:italic>\n                    ) localizes to human chromosome 10p15.3 and is associated with neurodevelopmental disorders. It encodes the protein DIP2C that contains both an acyl\u2010CoA synthetase domain and an AMP\u2010binding domain, which are involved in lipid metabolism. To investigate the role of DIP2C in neurodevelopment, we constructed both heterozygous and homozygous\n                    <jats:italic>Dip2c<\/jats:italic>\n                    mutant mice. Homozygous mutant mice exhibited weight loss, hyperlocomotion, cognitive impairment, and abnormal lipid metabolism, whereas heterozygous mutant mice displayed only mild cognitive impairment, recapitulating the dosage\u2010sensitive phenotype observed in human 10p15.3 microdeletion syndrome. Mendelian randomization analysis suggested a positive causal relationship between linoleic acid and developmental disorders. Pathway enrichment analysis revealed a significant upregulation of sphingolipid metabolism in the cortex, accompanied by elevated oligodendrogenesis and myelination. This effect was likely due to increased expression of a key subunit of serine palmitoyltransferase, the rate\u2010limiting enzyme in sphingolipid biosynthesis. Together, these findings identify DIP2C as a critical regulator of myelination and sphingolipid metabolic homeostasis during neurodevelopment, offering novel insight into neurodevelopmental disorders.\n                  <\/jats:p>","DOI":"10.1111\/nyas.70090","type":"journal-article","created":{"date-parts":[[2025,10,7]],"date-time":"2025-10-07T05:47:13Z","timestamp":1759816033000},"page":"437-448","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["DIP2C Deficiency Leads to Abnormal Sphingolipid Metabolism in Mice"],"prefix":"10.1111","volume":"1553","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9185-4962","authenticated-orcid":false,"given":"Weitai","family":"Chai","sequence":"first","affiliation":[{"name":"Key Laboratory of Molecular Epigenetics, Ministry of Education, School of Life Sciences, Provincial Key Laboratory of Neuroplasticity Northeast Normal University  Changchun 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