{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,9]],"date-time":"2025-11-09T07:00:53Z","timestamp":1762671653202},"reference-count":36,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2002,10,31]],"date-time":"2002-10-31T00:00:00Z","timestamp":1036022400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J of Cellular Biochemistry"],"published-print":{"date-parts":[[2003,2]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Mutations in the human 25\u2010hydroxyvitamin\u2010D<jats:sub>3<\/jats:sub>\u20101\u03b1\u2010hydroxylase (CYP27B1) gene cause pseudo vitamin D deficiency rickets (PDDR). The kidney is the main site of expression of the <jats:italic>CYP27B1<\/jats:italic> gene, but expression has been documented in other cell types, including chondrocytes. We engineered a tissue\u2010specific and a conventional knockout of CYP27B1 in mice. The conventional knockout strain reproduced the PDDR phenotype. Homozygote mutant animals were treated with 1,25(OH)<jats:sub>2<\/jats:sub>D<jats:sub>3<\/jats:sub> or fed a high\u2010calcium diet (2% calcium, 1.25% phosphate, 20% lactose) for 5 weeks post\u2010weaning. Blood biochemistry revealed that both rescue treatments corrected the hypocalcemia and secondary hyperparathyroidism. Bone histomorphometry confirmed that rickets were cured. The rescue regimen restored the biomechanical properties of the bone tissue. Mice carrying the loxP\u2010bearing allele were bred to transgenic animals expressing the Cre recombinase in chondrocytes under the control of the collagen type II promoter. Genotyping confirmed excision of exon 8 in chondrocytes. Serum biochemistry revealed that mineral ion homeostasis is normal in mutant animals. Preliminary observation of bone tissue from mutant mice did not reveal major changes to the growth plate. Precise histomorphometric analysis will be required to assess the impact of chondrocyte\u2010specific inactivation of CYP27B1 on the maturation and function of growth plate cells in vivo. J. Cell. Biochem. 88: 245\u2013251, 2003. \u00a9 2002 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/jcb.10348","type":"journal-article","created":{"date-parts":[[2003,1,9]],"date-time":"2003-01-09T21:06:13Z","timestamp":1042146373000},"page":"245-251","source":"Crossref","is-referenced-by-count":38,"title":["Conventional and tissue\u2010specific inactivation of the 25\u2010hydroxyvitamin D\u20101\u03b1\u2010hydroxylase (CYP27B1)"],"prefix":"10.1002","volume":"88","author":[{"given":"Ren\u00e9","family":"St\u2010Arnaud","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Olivier","family":"Dardenne","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jos\u00e9e","family":"Prud'homme","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"S. 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