{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,12]],"date-time":"2026-08-12T06:28:42Z","timestamp":1786516122021,"version":"build-2736575974"},"reference-count":37,"publisher":"Wiley","issue":"15","license":[{"start":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T00:00:00Z","timestamp":1785283200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T00:00:00Z","timestamp":1785283200000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["91439130"],"award-info":[{"award-number":["91439130"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82170235"],"award-info":[{"award-number":["82170235"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82470245"],"award-info":[{"award-number":["82470245"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82022018"],"award-info":[{"award-number":["82022018"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["32070958"],"award-info":[{"award-number":["32070958"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001809","name":"National Natural Science Foundation of China","doi-asserted-by":"publisher","award":["82161138025"],"award-info":[{"award-number":["82161138025"]}],"id":[{"id":"10.13039\/501100001809","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["faseb.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["The FASEB Journal"],"published-print":{"date-parts":[[2026,8,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    TANGO2 deficiency disorder (TDD) is a rare genetic disease caused by mutations in the\n                    <jats:italic>TANGO2<\/jats:italic>\n                    gene, characterized by prominent neurological symptoms. However, the pathological mechanisms underlying TANGO2 loss\u2010of\u2010function in neurologic symptoms remain unknown. Here, we generated constitutive and cell\u2010type\u2010specific\n                    <jats:italic>Tango2<\/jats:italic>\n                    knockout mouse models to examine TANGO2's role in the central nervous system (CNS). Behavioral analyses revealed that both constitutive and oligodendrocyte\u2010specific deletion of\n                    <jats:italic>Tango2<\/jats:italic>\n                    recapitulate\u00a0the motor deficits associated with individuals with TDD. Morphological quantifications further showed that\n                    <jats:italic>Tango2<\/jats:italic>\n                    deletion led to robust cerebellar myelin loss and an increase in synapse number in the cerebellar cortex. In addition, transcriptional analysis and lipidomic profiling demonstrated that\n                    <jats:italic>Tango2<\/jats:italic>\n                    deletion downregulated key processes involved in phospholipid metabolism. Significantly, vitamin B5 supplementation alleviated motor deficits and cerebellar myelin defects in\n                    <jats:italic>Tango2<\/jats:italic>\n                    knockout mice. Overall, our findings establish that TANGO2 is essential for maintaining normal motor behaviors by regulating lipid metabolism in oligodendroglia.\n                  <\/jats:p>","DOI":"10.1096\/fj.202601407rr","type":"journal-article","created":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T10:43:40Z","timestamp":1785321820000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Oligodendroglial\n                    <scp>TANGO2<\/scp>\n                    Regulates Lipid Metabolism to Control Motor Coordination"],"prefix":"10.1096","volume":"40","author":[{"ORCID":"https:\/\/orcid.org\/0009-0008-2079-4721","authenticated-orcid":false,"given":"Jiewen","family":"Chen","sequence":"first","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhili","family":"Liu","sequence":"additional","affiliation":[{"name":"Shanghai Key Laboratory of Emotions and Affective Disorders, Songjiang Research Institute Songjiang Hospital Affiliated to Shanghai Jiao Tong University School of Medicine  Shanghai China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fengling","family":"Chen","sequence":"additional","affiliation":[{"name":"BGI Research  Sanya China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yongfei","family":"Cui","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"},{"name":"Institute of Neurological and Psychiatric Disorders, Shenzhen Bay Laboratory  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liming","family":"Qin","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"},{"name":"Institute of Neurological and Psychiatric Disorders, Shenzhen Bay Laboratory  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mengdi","family":"Wang","sequence":"additional","affiliation":[{"name":"School of Life Sciences Mudanjiang Medical University  Mudanjiang China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiangbin","family":"Zhu","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Na","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Can","family":"Huang","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bo","family":"Zhang","sequence":"additional","affiliation":[{"name":"Institute of Neurological and Psychiatric Disorders, Shenzhen Bay Laboratory  Shenzhen China"},{"name":"Shenzhen Medical Academy of Research and Translation (SMART)  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0292-375X","authenticated-orcid":false,"given":"Kunfu","family":"Ouyang","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Surgery, Peking University Shenzhen Hospital, School of Chemical Biology and Biotechnology Peking University Shenzhen Graduate School, Peking University  Shenzhen China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,29]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.gim.2022.11.020"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.62543"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/jimd.12314"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2015.12.009"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/jimd.12149"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature04377"},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.7554\/eLife.85345"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1083\/jcb.202410001"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/jimd.12312"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-022-07076-9"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/jimd.12156"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467-025-60563-1"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1242\/dmm.050092"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbrc.2024.150047"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.63938"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1515\/jpem-2023-0172"},{"key":"e_1_2_11_18_1","article-title":"Myelin Lipid Metabolism and Its Role in Myelination and Myelin Maintenance","volume":"4","author":"Barnes\u2010V\u00e9lez J. A.","year":"2023","journal-title":"Innovation"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1194\/jlr.R009761"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2345-24.2025"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1038\/nprot.2006.116"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41586-022-04722-0"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.arcped.2020.11.004"},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2015.12.008"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awn157"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.22337"},{"key":"e_1_2_11_27_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41583-023-00744-3"},{"key":"e_1_2_11_28_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41380-024-02659-6"},{"key":"e_1_2_11_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2015.07.020"},{"key":"e_1_2_11_30_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.1356-16.2016"},{"key":"e_1_2_11_31_1","doi-asserted-by":"publisher","DOI":"10.1002\/jimd.12579"},{"key":"e_1_2_11_32_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41583-020-00379-8"},{"key":"e_1_2_11_33_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrneurol.2017.175"},{"key":"e_1_2_11_34_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00401-017-1739-1"},{"key":"e_1_2_11_35_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2010.09.009"},{"key":"e_1_2_11_36_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.047951"},{"key":"e_1_2_11_37_1","doi-asserted-by":"publisher","DOI":"10.1111\/jns.12625"},{"key":"e_1_2_11_38_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrn3484"}],"container-title":["The FASEB Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/faseb.onlinelibrary.wiley.com\/doi\/pdf\/10.1096\/fj.202601407RR","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/faseb.onlinelibrary.wiley.com\/doi\/full-xml\/10.1096\/fj.202601407RR","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/faseb.onlinelibrary.wiley.com\/doi\/pdf\/10.1096\/fj.202601407RR","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,12]],"date-time":"2026-08-12T05:42:01Z","timestamp":1786513321000},"score":1,"resource":{"primary":{"URL":"https:\/\/faseb.onlinelibrary.wiley.com\/doi\/10.1096\/fj.202601407RR"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,7,29]]},"references-count":37,"journal-issue":{"issue":"15","published-print":{"date-parts":[[2026,8,15]]}},"alternative-id":["10.1096\/fj.202601407RR"],"URL":"https:\/\/doi.org\/10.1096\/fj.202601407rr","archive":["Portico"],"relation":{},"ISSN":["0892-6638","1530-6860"],"issn-type":[{"value":"0892-6638","type":"print"},{"value":"1530-6860","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,7,29]]},"assertion":[{"value":"2026-03-23","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-16","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-29","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e72155"}}