{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,24]],"date-time":"2026-07-24T06:54:27Z","timestamp":1784876067947,"version":"3.55.0"},"reference-count":40,"publisher":"The Endocrine Society","issue":"11","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2014,11,1]]},"abstract":"<jats:sec>\n                    <jats:title>Context:<\/jats:title>\n                    <jats:p>Fibrous dysplasia (FD) is a rare skeletal disorder, resulting in deformity, fracture, functional impairment, and pain. Bisphosphonates have been advocated as a potential treatment.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective:<\/jats:title>\n                    <jats:p>To determine the efficacy of alendronate for treatment of FD.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design:<\/jats:title>\n                    <jats:p>Two-year randomized, double-blind, placebo-controlled trial.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Setting:<\/jats:title>\n                    <jats:p>Clinical research center.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Patients:<\/jats:title>\n                    <jats:p>Forty subjects with polyostotic FD (24 adults, 16 children). Subjects were randomized and stratified by age.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interventions:<\/jats:title>\n                    <jats:p>Study drug was administered over a 24 month period in 6 month cycles (6 months on, 6 months off). Alendronate dosing was stratified: 40 mg daily for subjects &amp;gt;50 kg, 20 mg for 30\u201350 kg, 10 mg for 20\u201330 kg.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Main Outcome Measures:<\/jats:title>\n                    <jats:p>Primary endpoints were bone turnover markers, including serum osteocalcin, and urinary NTX-telopeptides. Secondary endpoints included areal bone mineral density (aBMD), pain, skeletal disease burden score, and functional parameters including the 9-min walk test and manual muscle testing.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>Clinical data was collected on 35 subjects who completed the study. There was a decline in NTX-telopeptides in the alendronate group (P = .006), but no significant difference in osteocalcin between groups. The alendronate group had an increase in areal BMD in normal bone at the lumbar spine (P = .006), and in predetermined regions of FD (P &amp;lt; .001). There were no significant differences in pain scores, skeletal disease burden scores, or functional parameters between the groups.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>Alendronate treatment led to a reduction in the bone resorption marker NTX-telopeptides, and improvement in aBMD, but no significant effect on serum osteocalcin, pain, or functional parameters.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1210\/jc.2014-1371","type":"journal-article","created":{"date-parts":[[2014,7,17]],"date-time":"2014-07-17T11:51:50Z","timestamp":1405597910000},"page":"4133-4140","source":"Crossref","is-referenced-by-count":132,"title":["A Randomized, Double Blind, Placebo-Controlled Trial of Alendronate Treatment for Fibrous Dysplasia of Bone"],"prefix":"10.1210","volume":"99","author":[{"given":"Alison M.","family":"Boyce","sequence":"first","affiliation":[{"name":"Skeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch (A.M.B., M.H.K., B.A.B., P.G.R., M.T.C.), National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland 20892"},{"name":"Division of Endocrinology and Diabetes (A.M.B.), Children's National Health System, Washington, DC 20010"},{"name":"Bone Health Program, Division of Orthopaedics and Sports Medicine (A.M.B.), Children's National Health System, Washington, DC 20010"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marilyn H.","family":"Kelly","sequence":"additional","affiliation":[{"name":"Skeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch (A.M.B., M.H.K., B.A.B., P.G.R., M.T.C.), National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland 20892"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beth A.","family":"Brillante","sequence":"additional","affiliation":[{"name":"Skeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch (A.M.B., M.H.K., B.A.B., P.G.R., M.T.C.), National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland 20892"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harvey","family":"Kushner","sequence":"additional","affiliation":[{"name":"BioMedical Computer Research Institute (H.K.), Philadelphia, Pennsylvania 19115"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shlomo","family":"Wientroub","sequence":"additional","affiliation":[{"name":"Department of Pediatric Orthopedics (S.W.), Dana Children's Hospital, Tel-Aviv Sourasky Medical Center, Sackler Faculty of Medicine, Tel-Aviv University, Israel 64239"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mara","family":"Riminucci","sequence":"additional","affiliation":[{"name":"Department of Molecular Medicine (M.R., P.B.), La Sapienza Universita di Roma, Rome, Italy 00185"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paolo","family":"Bianco","sequence":"additional","affiliation":[{"name":"Department of Molecular Medicine (M.R., P.B.), La Sapienza Universita di Roma, Rome, Italy 00185"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pamela G.","family":"Robey","sequence":"additional","affiliation":[{"name":"Skeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch (A.M.B., M.H.K., B.A.B., P.G.R., M.T.C.), National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland 20892"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael T.","family":"Collins","sequence":"additional","affiliation":[{"name":"Skeletal Clinical Studies Unit, Craniofacial and Skeletal Diseases Branch (A.M.B., M.H.K., B.A.B., P.G.R., M.T.C.), National Institute of Dental and Craniofacial Research, 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