{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,30]],"date-time":"2026-07-30T04:35:14Z","timestamp":1785386114955,"version":"3.55.0"},"reference-count":127,"publisher":"The Company of Biologists","issue":"2","license":[{"start":{"date-parts":[[2025,1,20]],"date-time":"2025-01-20T00:00:00Z","timestamp":1737331200000},"content-version":"vor","delay-in-days":5,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100007172","name":"Cincinnati Children's Hospital Medical Center","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007172","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Cincinnati Children's Research Foundation"},{"DOI":"10.13039\/501100000268","name":"Biotechnology and Biological Sciences Research Council","doi-asserted-by":"crossref","award":["BB\/S015906\/1"],"award-info":[{"award-number":["BB\/S015906\/1"]}],"id":[{"id":"10.13039\/501100000268","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100008102","name":"University of Cincinnati","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100008102","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,1,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Heterozygous variants in SOX10 cause congenital syndromes affecting pigmentation, digestion, hearing, and neural development, primarily attributable to failed differentiation or loss of non-skeletal neural crest derivatives. We report here an additional, previously undescribed requirement for Sox10 in bone mineralization. Neither crest- nor mesoderm-derived bones initiate mineralization on time in zebrafish sox10 mutants, despite normal osteoblast differentiation and matrix production. Mutants are deficient in the Trpv6+ ionocytes that take up calcium from the environment, resulting in severe calcium deficiency. As these ionocytes derive from ectoderm, not crest, we hypothesized that the primary defect resides in a separate organ that systemically regulates ionocyte numbers. RNA sequencing revealed significantly elevated stanniocalcin (Stc1a), an anti-hypercalcemic hormone, in sox10 mutants. Stc1a inhibits calcium uptake in fish by repressing trpv6 expression and Trpv6+ ionocyte proliferation. Epistasis assays confirm excess Stc1a as the proximate cause of the calcium deficit. The pronephros-derived glands that synthesize Stc1a interact with sox10+ cells, but these cells are missing in mutants. We conclude that sox10+ crest-derived cells non-autonomously limit Stc1a production to allow the inaugural wave of calcium uptake necessary to initiate bone mineralization.<\/jats:p>","DOI":"10.1242\/dev.204357","type":"journal-article","created":{"date-parts":[[2025,1,10]],"date-time":"2025-01-10T09:26:32Z","timestamp":1736501192000},"source":"Crossref","is-referenced-by-count":8,"title":["Sox10 is required for systemic initiation of bone mineralization"],"prefix":"10.1242","volume":"152","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-6835-8315","authenticated-orcid":false,"given":"Stefani","family":"Gjorcheska","sequence":"first","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Human Genetics , , Cincinnati, OH 45229,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandhya","family":"Paudel","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Human Genetics , , Cincinnati, OH 45229,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sarah","family":"McLeod","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Human Genetics , , Cincinnati, OH 45229,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7692-7924","authenticated-orcid":false,"given":"David","family":"Paulding","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Human Genetics , , Cincinnati, OH 45229,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Louisa","family":"Snape","sequence":"additional","affiliation":[{"name":"University of Bath 2 Department of Life Sciences , , Bath BA2 7AY ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karen Camargo","family":"Sosa","sequence":"additional","affiliation":[{"name":"University of Bath 2 Department of Life Sciences , , Bath BA2 7AY ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6794-2762","authenticated-orcid":false,"given":"Cunming","family":"Duan","sequence":"additional","affiliation":[{"name":"University of Michigan 3 Department of Molecular, Cellular, and Developmental Biology , , Ann Arbor, MI 48109,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9381-0066","authenticated-orcid":false,"given":"Robert","family":"Kelsh","sequence":"additional","affiliation":[{"name":"University of Bath 2 Department of Life Sciences , , Bath BA2 7AY ,","place":["UK"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1275-7978","authenticated-orcid":false,"given":"Lindsey","family":"Barske","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center 1 Division of Human Genetics , , Cincinnati, OH 45229,","place":["USA"]},{"name":"University of Cincinnati College of Medicine 4 Department of Pediatrics , , Cincinnati, OH 45229,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2025,1,20]]},"reference":[{"key":"2025012011254167800_DEV204357C1","doi-asserted-by":"publisher","first-page":"267","DOI":"10.1002\/cne.10214","article-title":"Differentiation and maturation of zebrafish dorsal root and sympathetic ganglion neurons","volume":"446","author":"An","year":"2002","journal-title":"J. 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