{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T14:39:19Z","timestamp":1786977559631,"version":"build-2736575974"},"reference-count":37,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2022,2,18]],"date-time":"2022-02-18T00:00:00Z","timestamp":1645142400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["DE025897"],"award-info":[{"award-number":["DE025897"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100008732","name":"Uehara Memorial Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100008732","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2022,7]]},"abstract":"Abstract<\/jats:title>\n \n Background<\/jats:title>\n \n Absence of Golgi microtubule\u2010associated protein 210 (GMAP210), encoded by the\n TRIP11<\/jats:italic>\n gene, results in achondrogenesis. Although\n TRIP11<\/jats:italic>\n is thought to be specifically required for chondrogenesis, human fetuses with the mutation of\n TRIP11<\/jats:italic>\n also display bony skull defects where chondrocytes are usually not present. This raises an important question of how TRIP11 functions in bony skull development.\n <\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n We disrupted\n Trip11<\/jats:italic>\n in neural crest\u2010derived cell populations, which are critical for developing skull in mice. In\n Trip11<\/jats:italic>\n mutant skulls, expression levels of ER stress markers were increased compared to controls. Morphological analysis of electron microscopy data revealed swollen ER in\n Trip11<\/jats:italic>\n mutant skulls. Unexpectedly, we also found that Golgi stress increased in\n Trip11<\/jats:italic>\n mutant skulls, suggesting that both ER and Golgi stress\u2010induced cell death may lead to osteopenia\u2010like phenotypes in\n Trip11<\/jats:italic>\n mutant skulls. These data suggest that\n Trip11<\/jats:italic>\n plays pivotal roles in the regulation of ER and Golgi stress, which are critical for osteogenic cell survival.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusion<\/jats:title>\n \n We have recently reported that the molecular complex of ciliary protein and GMAP210 is required for collagen trafficking. In this paper, we further characterized the important role of\n Trip11<\/jats:italic>\n being possibly involved in the regulation of ER and Golgi stress during skull development.\n <\/jats:p>\n <\/jats:sec>","DOI":"10.1002\/dvdy.461","type":"journal-article","created":{"date-parts":[[2022,2,11]],"date-time":"2022-02-11T05:54:04Z","timestamp":1644558844000},"page":"1209-1222","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Disruption of\n Trip11<\/i>\n in cranial neural crest cells is associated with increased\n ER<\/scp>\n and Golgi stress contributing to skull defects in mice"],"prefix":"10.1002","volume":"251","author":[{"given":"Hiroyuki","family":"Yamaguchi","sequence":"first","affiliation":[{"name":"Department of Pediatrics McGovern Medical School, The University of Texas Health Science Center at Houston Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Matthew D.","family":"Meyer","sequence":"additional","affiliation":[{"name":"Shared Equipment Authority Rice University Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Li","family":"He","sequence":"additional","affiliation":[{"name":"Department of Pediatrics McGovern Medical School, The University of Texas Health Science Center at Houston Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3115-1437","authenticated-orcid":false,"given":"Yoshihiro","family":"Komatsu","sequence":"additional","affiliation":[{"name":"Department of Pediatrics McGovern Medical School, The University of Texas Health Science Center at Houston Houston Texas USA"},{"name":"Graduate Program in Genetics and Epigenetics The University of Texas MD Anderson Cancer Center UTHealth Graduate School of Biomedical Sciences Houston Texas 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