{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,15]],"date-time":"2026-09-15T04:44:26Z","timestamp":1789447466289,"version":"build-2803163510"},"reference-count":23,"publisher":"Oxford University Press (OUP)","issue":"1","license":[{"start":{"date-parts":[[1993,1,1]],"date-time":"1993-01-01T00:00:00Z","timestamp":725846400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"DOI":"10.13039\/501100007155","name":"Medical Research Council of Canada","doi-asserted-by":"crossref","award":["MT4515"],"award-info":[{"award-number":["MT4515"]}],"id":[{"id":"10.13039\/501100007155","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100007155","name":"Medical Research Council of Canada","doi-asserted-by":"crossref","award":["MA10074"],"award-info":[{"award-number":["MA10074"]}],"id":[{"id":"10.13039\/501100007155","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"U.S. National Institutes of Health","doi-asserted-by":"crossref","award":["AM34281"],"award-info":[{"award-number":["AM34281"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[1993,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>A young girl had tibial osteotomies at age 14 for genu valgum and then had recurrent tibia) cysts over a number of years. Hypocalcemia and hyperphosphatemia were first noted at age 21. The diagnosis of pseudohypoparathyroidism was made at age 28, when elevated plasma PTH was detected. Clinical and biochemical features, including a PTH response test and assay of RBC Gs, established the diagnosis of pseudohypoparathyroidism type lb. Failure to suppress plasma PTH with vitamin D therapy led to an exacerbation of her cystic bone disease; there were widespread lytic lesions radiologically, most of which took up [99mTc]diphosphonate on bone scan. Microradioscopy revealed evidence of resorption of phalangeal tufts. Bone biopsy showed osteitis fibrosa cystica. During an orthopedic procedure, trabecular bone fragments were taken from her right humerus, and bone-derived cells cultured using an explant technique. The cultured cells were osteoblast-like in morphology, fully responsive to PTH, cholera toxin, forskolin, and PGE1 in vitro, and had an alkaline phosphatase and osteocalcin response to 1,25-dihydroxyvitamin D3 [1,25-(OH)2D3]. Following this examination of skeletal responsiveness, attempts were made to suppress the elevated plasma PTH levels and symptomatic bone disease by optimizing therapy with oral 1,25-(OH)2D3. When bone pain associated with the cystic bone disease failed to resolve, the patient underwent total parathyroidectomy, following which the bone pain gradually resolved. This is the first direct demonstration of PTH responsiveness in cultured bone cells in the syndrome of pseudohypoparathyroidism with osteitis fibrosa cystica.<\/jats:p>","DOI":"10.1002\/jbmr.5650080111","type":"journal-article","created":{"date-parts":[[2010,2,3]],"date-time":"2010-02-03T11:44:27Z","timestamp":1265197467000},"page":"83-91","source":"Crossref","is-referenced-by-count":74,"title":["Pseudohypoparathyroidism with osteitis fibrosa cystica: Direct demonstration of skeletal responsiveness to parathyroid hormone in cells cultured from bone"],"prefix":"10.1093","volume":"8","author":[{"given":"Timothy M.","family":"Murray","sequence":"first","affiliation":[{"name":"Division of Endocrinology and Metabolism, St. Michael's Hospital, and the Department of Medicine, University of Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leticia Gomez","family":"Rao","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism, St. Michael's Hospital, and the Department of Medicine, University of Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Min-Min","family":"Wong","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism, St. Michael's Hospital, and the Department of Medicine, University of Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James P.","family":"Waddell","sequence":"additional","affiliation":[{"name":"Departments of Surgery, St. Michael's Hospital and University of Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert","family":"McBroom","sequence":"additional","affiliation":[{"name":"Departments of Surgery, St. Michael's Hospital and University of Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cherk S.","family":"Tam","sequence":"additional","affiliation":[{"name":"Department of Medicine, University of Toronto, and the Research Institute, Queen Elizabeth Hospital, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fred","family":"Rosen","sequence":"additional","affiliation":[{"name":"Scarborough General Hospital, Toronto, Ontario, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael A.","family":"Levine","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Johns Hopkins University School of Medicine, Baltimore, Maryland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[1993,1,1]]},"reference":[{"key":"2024013113121999800_bib1","author":"Murray","year":"1985"},{"key":"2024013113121999800_bib2","first-page":"922","article-title":"Pseudohypoparathyroidism: An example of \u201cSeabright-Bantam syndrome.\u201d","volume":"30","author":"Albright","year":"1942","journal-title":"Endocrinology"},{"key":"2024013113121999800_bib3","doi-asserted-by":"crossref","first-page":"1832","DOI":"10.1172\/JCI106149","article-title":"Pseudohypoparathyroidism: Defective excretion of 3\u2032,5\u2032-AMP in response to parathyroid hormone","volume":"48","author":"Chase","year":"1969","journal-title":"J Clin Invest"},{"key":"2024013113121999800_bib4","doi-asserted-by":"crossref","first-page":"1319","DOI":"10.1016\/0006-291X(80)90563-X","article-title":"Deficient activity of guanine nucleotide regulatory protein in erythrocytes from patients with pseudohypoparathyroidism","volume":"94","author":"Levine","year":"1980","journal-title":"Biochem Biophys Res Commun"},{"key":"2024013113121999800_bib5","doi-asserted-by":"crossref","first-page":"545","DOI":"10.1016\/0002-9343(83)91008-2","article-title":"Resistance to multiple hormones in patients with pseudohypoparathyroidism: Association with deficient activity of guanine nucleotide regulatory protein","volume":"74","author":"Levine","year":"1983","journal-title":"Am J Med"},{"key":"2024013113121999800_bib6","doi-asserted-by":"crossref","first-page":"493","DOI":"10.1677\/joe.0.0560493","article-title":"The immunochemical specificity of antisera to bovine parathyroid hormone: An approach to region-specific immunoassay","volume":"56","author":"Murray","year":"1973","journal-title":"J Endocrinol"},{"key":"2024013113121999800_bib7","doi-asserted-by":"crossref","first-page":"287","DOI":"10.1093\/clinchem\/24.2.287","article-title":"Assay of vitamins D2 and D3 and 25-hydroxy-vitamins D2 and D3 in human plasma by high-performance liquid chromatography","volume":"24","author":"Jones","year":"1978","journal-title":"Clin Chem"},{"key":"2024013113121999800_bib8","doi-asserted-by":"crossref","first-page":"38","DOI":"10.1042\/cs064038Pb","article-title":"Human bone cells in culture. 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