{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,26]],"date-time":"2025-10-26T22:46:58Z","timestamp":1761518818191},"reference-count":20,"publisher":"Oxford University Press (OUP)","issue":"9","license":[{"start":{"date-parts":[[2001,9,1]],"date-time":"2001-09-01T00:00:00Z","timestamp":999302400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2001,9,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Bisphosphonate treatment for severe Paget's disease leads to hypocalcemia followed by a secondary hyperparathyroid response to restore normocalcemia. A case is presented of a 60-year-old woman with polyostotic Paget's disease and postsurgical hypoparathyroidism. In 1993 her Paget's disease\u2014alkaline phosphatase (ALP), 1260 U\/liter (35\u2013135 U\/liter), and fasting urinary hydroxyproline excretion, 13.7 \u03bcmol\/liter GF (0.4\u20131.9 \u03bcmol\/liter)\u2014was treated with intravenous pamidronate. Symptomatic hypocalcemia followed the first 60-mg dose, requiring large doses of calcium supplementation and calcitriol. Pamidronate therapy to a total dose of 360 mg was followed by rapid and prolonged remission with indices of bone turnover in the normal range within 2 months and persisting for at least 19 months after treatment. In 1999 relapse of Paget's disease\u2014ALP, 511 U\/liter (35\u2013135 U\/liter), and fasting urinary deoxypyridinoline\/creatinine 53.1 \u03bcmol\/mol (5\u201327 \u03bcmol\/mol)\u2014was treated with alendronate, 10 mg daily. Symptomatic hypocalcemia occurred again, requiring increased calcium and calcitriol therapy. Indices of bone turnover were within the normal range 9 weeks after the start of therapy. These responses were significantly more rapid and sustained than those observed in euparathyroid subjects. This case suggests that the lack of parathyroid response may modify the response to bisphosphonates by: (a) increasing intrinsic uptake of bisphosphonate into the pagetic skeleton, allowing response to a smaller dose; (b) increasing duration and severity of hypocalcemia after bisphosphonate therapy; and (c) removing the hyperparathyroid drive to reactivation of pagetic osteoclasts, leading to a prolonged remission. These observations have implications for optimizing bisphosphonate therapy both in Paget's disease and in osteoporosis.<\/jats:p>","DOI":"10.1359\/jbmr.2001.16.9.1719","type":"journal-article","created":{"date-parts":[[2006,4,26]],"date-time":"2006-04-26T20:52:32Z","timestamp":1146084752000},"page":"1719-1723","source":"Crossref","is-referenced-by-count":13,"title":["Bisphosphonate Therapy for Paget's Disease in a Patient with Hypoparathyroidism: Profound Hypocalcemia, Rapid Response, and Prolonged Remission"],"prefix":"10.1093","volume":"16","author":[{"given":"B. G. A.","family":"Stuckey","sequence":"first","affiliation":[{"name":"Department of Endocrinology and Diabetes, Sir Charles Gairdner Hospital, QEII Medical Center, Nedlands, Western Australia, Australia"},{"name":"Keogh Institute for Medical Research, QEII Medical Center, Nedlands, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"E. M.","family":"Lim","sequence":"additional","affiliation":[{"name":"Division of Clinical Pathology, PathCenter, QEII Medical Center, Nedlands, Western Australia, Australia"},{"name":"Department of Endocrinology and Diabetes, Sir Charles Gairdner Hospital, QEII Medical Center, Nedlands, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"G. N.","family":"Kent","sequence":"additional","affiliation":[{"name":"Division of Clinical Pathology, PathCenter, QEII Medical Center, Nedlands, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"L. C.","family":"Ward","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Diabetes, Sir Charles Gairdner Hospital, QEII Medical Center, Nedlands, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"D. H.","family":"Gutteridge","sequence":"additional","affiliation":[{"name":"Department of Endocrinology and Diabetes, Sir Charles Gairdner Hospital, QEII Medical Center, Nedlands, Western Australia, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2009,12,2]]},"reference":[{"key":"2024020714123106100_bib1","doi-asserted-by":"crossref","first-page":"1012","DOI":"10.1136\/ard.45.12.1012","article-title":"Low dose intravenous 3-amino-1-hydroxypropylidene-1,1-bisphosphonate (APD) for the treatment of Paget's disease of bone","volume":"45","author":"Cantrill","year":"1986","journal-title":"Ann Rheum Dis"},{"key":"2024020714123106100_bib2","first-page":"69","article-title":"Effects of decreasing serum calcium on circulating parathyroid hormone and vitamin D metabolites in normocalcaemic and hypercalcaemic patients treated with APD","volume":"1","author":"Papapoulos","year":"1986","journal-title":"Bone Miner"},{"key":"2024020714123106100_bib3","doi-asserted-by":"crossref","first-page":"1100","DOI":"10.1210\/jcem-55-6-1100","article-title":"Secondary hyperparathyroidism associated with dichloromethane diphosphonate treatment of Paget's disease","volume":"55","author":"Harris","year":"1982","journal-title":"J Clin Endocrinol Metab"},{"key":"2024020714123106100_bib4","doi-asserted-by":"crossref","first-page":"837","DOI":"10.1210\/jcem-54-4-837","article-title":"Long term effects of dichloromethylene diphosphonate in Paget's disease of bone","volume":"54","author":"Delmas","year":"1982","journal-title":"J Clin Endocrinol Metab"},{"key":"2024020714123106100_bib5","doi-asserted-by":"crossref","first-page":"387","DOI":"10.1016\/S8756-3282(96)00224-4","article-title":"Clinical, biochemical, hematologic and radiographic responses in Paget's disease following intravenous pamidronate disodium: A 2-year study","volume":"19","author":"Gutteridge","year":"1996","journal-title":"Bone"},{"key":"2024020714123106100_bib6","doi-asserted-by":"crossref","first-page":"693","DOI":"10.1210\/jcem-35-5-693","article-title":"Hypoparathyroidism and Paget's disease: The effect of parathyroid administration","volume":"35","author":"Genuth","year":"1972","journal-title":"J Clin Endocrinol"},{"key":"2024020714123106100_bib7","doi-asserted-by":"crossref","first-page":"286","DOI":"10.1097\/00000658-196908000-00019","article-title":"Osteitis deformans (Paget's disease of bone) complicating hypoparathyroidism","volume":"170","author":"Pitman","year":"1969","journal-title":"Ann Surg"},{"key":"2024020714123106100_bib8","doi-asserted-by":"crossref","first-page":"407","DOI":"10.1016\/S0936-6555(98)80045-9","article-title":"Severe prolonged hypocalcaemia following pamidronate for malignant hypercalcaemia","volume":"10","author":"Sims","year":"1998","journal-title":"Clin Oncol"},{"key":"2024020714123106100_bib9","doi-asserted-by":"crossref","first-page":"396","DOI":"10.2169\/internalmedicine.37.396","article-title":"Symptomatic hypocalcemia in a patient with latent hypoparathyroidism and breast carcinoma with bone metastasis following administration of pamidronate","volume":"37","author":"Comlekci","year":"1998","journal-title":"Intern Med"},{"key":"2024020714123106100_bib10","first-page":"800","article-title":"Uptake of alendronate by bone tissue in hypocalcemic and hypercalcemic rats","volume":"21","author":"Lin","year":"1993","journal-title":"Drug Metab Dispos"},{"key":"2024020714123106100_bib11","doi-asserted-by":"crossref","first-page":"209","DOI":"10.1002\/jbmr.5650080212","article-title":"Rapid, divergent changes in spinal and forearm bone density following short-term intravenous treatment of Paget's disease with pamidronate disodium","volume":"8","author":"Price","year":"1993","journal-title":"J Bone Miner Res"},{"key":"2024020714123106100_bib12","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1016\/S8756-3282(98)00163-X","article-title":"Prevention of appendicular bone loss in Paget's disease following treatment with intravenous pamidronate","volume":"24","author":"Stewart","year":"1999","journal-title":"Bone"},{"key":"2024020714123106100_bib13","doi-asserted-by":"crossref","first-page":"301","DOI":"10.1016\/0221-8747(81)90046-1","article-title":"Clinical and biochemical effects of Paget's disease of bone: Patterns of response to initial treatment and to long-term therapy","volume":"45","author":"Siris","year":"1981","journal-title":"Metab Bone Dis Relat Res"},{"key":"2024020714123106100_bib14","first-page":"30","author":"Stuckey","year":"1995"},{"key":"2024020714123106100_bib15","first-page":"231","article-title":"Technetium-99m-pyrophosphate bone scans in hyperparathyroidism","volume":"18","author":"Wiegmann","year":"1977","journal-title":"J Nucl Med"},{"key":"2024020714123106100_bib16","doi-asserted-by":"crossref","first-page":"611","DOI":"10.3109\/00365518809085780","article-title":"Non-invasive evaluation of bone formation: Measurements of serum alkaline phosphatase, whole body retention of diphosphonate and serum osteocalcin in metabolic bone disorders and thyroid disease","volume":"48","author":"Hyldstrup","year":"1988","journal-title":"Scand J Clin Lab Invest"},{"key":"2024020714123106100_bib17","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1016\/8756-3282(94)90818-4","article-title":"Effects of two oral doses of alendronate in the treatment of Paget's disease of bone","volume":"15","author":"Adami","year":"1994","journal-title":"Bone"},{"key":"2024020714123106100_bib18","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1016\/S8756-3282(96)00364-X","article-title":"Alendronate in the treatment of Paget's disease of bone","volume":"20","author":"Khan","year":"1997","journal-title":"Bone"},{"key":"2024020714123106100_bib19","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1002\/jbmr.5650140216","article-title":"Paget's disease: Acquired resistance to one aminobisphosphonate with retained response to another","volume":"14","author":"Gutteridge","year":"1999","journal-title":"J Bone Miner Res"},{"key":"2024020714123106100_bib20","doi-asserted-by":"crossref","first-page":"51","DOI":"10.1016\/S0169-6009(08)80186-9","article-title":"The effect of alendronate on renal tubular reabsorption of phosphate","volume":"27","author":"Vasikaran","year":"1994","journal-title":"Bone Miner"}],"container-title":["Journal of Bone and Mineral Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1359%2Fjbmr.2001.16.9.1719","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/16\/9\/1719\/56605259\/5650160919.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jbmr\/article-pdf\/16\/9\/1719\/56605259\/5650160919.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,3,13]],"date-time":"2024-03-13T15:03:33Z","timestamp":1710342213000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jbmr\/article\/16\/9\/1719-1723\/7515935"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2001,9,1]]},"references-count":20,"journal-issue":{"issue":"9","published-online":{"date-parts":[[2009,12,2]]},"published-print":{"date-parts":[[2001,9,1]]}},"URL":"https:\/\/doi.org\/10.1359\/jbmr.2001.16.9.1719","relation":{},"ISSN":["0884-0431","1523-4681"],"issn-type":[{"value":"0884-0431","type":"print"},{"value":"1523-4681","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2001,9,1]]},"published":{"date-parts":[[2001,9,1]]}}}