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Alternatively, they may arise from disrupted crosstalk among tissues. Here we show the local reduction of two vestibulo-cerebellar lobules, the paraflocculus and flocculus, in mouse models and humans with 22q11.2 deletion syndrome (22q11DS). In mice, this paraflocculus\/flocculus dysplasia is associated with haploinsufficiency of the\n                    <jats:italic>Tbx1<\/jats:italic>\n                    gene.\n                    <jats:italic>Tbx1<\/jats:italic>\n                    haploinsufficiency also leads to impaired cerebellar synaptic plasticity and motor learning. However, neural cell compositions and neurogenesis are not altered in the dysplastic paraflocculus\/flocculus. Interestingly, 22q11DS and\n                    <jats:italic>Tbx1<\/jats:italic>\n                    <jats:sup>\n                      <jats:italic>+\/\u2013<\/jats:italic>\n                    <\/jats:sup>\n                    mice have malformations of the subarcuate fossa, a part of the petrous temporal bone, which encapsulates the paraflocculus\/flocculus. Single-nuclei RNA sequencing reveals that\n                    <jats:italic>Tbx1<\/jats:italic>\n                    haploinsufficiency leads to precocious differentiation of chondrocytes to osteoblasts in the petrous temporal bone autonomous to paraflocculus\/flocculus cell populations. These findings suggest a previously unrecognized pathogenic structure\/function relation in 22q11DS in which local skeletal deformity and cerebellar dysplasia result in behavioral deficiencies.\n                  <\/jats:p>","DOI":"10.1038\/s41467-024-54837-3","type":"journal-article","created":{"date-parts":[[2024,12,5]],"date-time":"2024-12-05T05:02:18Z","timestamp":1733374938000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Tbx1 haploinsufficiency leads to local skull deformity, paraflocculus and flocculus dysplasia, and motor-learning deficit in 22q11.2 deletion syndrome"],"prefix":"10.1038","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7049-9079","authenticated-orcid":false,"given":"Tae-Yeon","family":"Eom","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7487-227X","authenticated-orcid":false,"given":"J. 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