{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,22]],"date-time":"2026-09-22T23:50:56Z","timestamp":1790121056471,"version":"4.0.1"},"reference-count":57,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2016,9,20]],"date-time":"2016-09-20T00:00:00Z","timestamp":1474329600000},"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":[[2017,2,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:sec>\n                  <jats:title>\u2003<\/jats:title>\n                  <jats:p>McCune-Albright syndrome (MAS) is a rare bone disorder characterized by fibrous dysplasia (FD), endocrinopathies, and caf\u00e9-au-lait patches. FD patients have been shown to respond favorably to treatment with bisphosphonates, but data are scarce in the more severe polyostotic form (PFD), including MAS, and factors determining treatment outcome are not known, particularly in the long-term. We evaluated the biochemical (bone turnover markers [BTMs]) and clinical (pain reduction) outcome of bisphosphonate therapy in 11 patients with MAS and 30 patients with PFD: median duration of treatment 6 years (range, 2 to 25 years). Prognostic factors for treatment outcome were identified in both groups. Patients with MAS were younger at diagnosis (p\u2009=\u20090.001), all had precocious puberty, and four (36%) had additional growth hormone (GH) excess associated with severe craniofacial FD. Extent of skeletal disease was more severe in MAS compared to PFD. MAS patients had higher serum alkaline phosphatase (ALP) concentrations (p\u2009=\u20090.005), higher skeletal burden scores (p\u2009&amp;lt;\u20090.001), and more fractures (p\u2009=\u20090.021). MAS patients had also higher levels of FGF-23 (p\u2009=\u20090.008) and higher prevalence of hypophosphatemia (p\u2009=\u20090.013). Twenty-four of 30 PFD patients (80%) demonstrated a complete clinical and biochemical response within a year of starting treatment (p\u2009=\u20090.015), compared to only four of 11 MAS patients (36%). There were no nonresponders. In the whole group, FGF-23, total ALP, P1NP, and CTX positively correlated with skeletal burden scores (all p\u2009\u2264\u20090.001), which was the only significant risk factor for an incomplete response to bisphosphonate therapy (p\u2009&amp;lt;\u20090.01). Our data suggest a beneficial and safe outcome of long-term bisphosphonate therapy in the majority of patients with PFD, although response to therapy was limited by the higher skeletal disease burden in MAS patients. In the PFD\/MAS population studied, the only identified prognostic factor that influenced the outcome of bisphosphonate therapy was a high skeletal burden score. \u00a9 2016 American Society for Bone and Mineral Research.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1002\/jbmr.2999","type":"journal-article","created":{"date-parts":[[2016,9,20]],"date-time":"2016-09-20T19:12:44Z","timestamp":1474398764000},"page":"264-276","source":"Crossref","is-referenced-by-count":86,"title":["Outcome of Long-Term Bisphosphonate Therapy in McCune-Albright Syndrome and Polyostotic Fibrous Dysplasia"],"prefix":"10.1093","volume":"32","author":[{"given":"Bas CJ","family":"Majoor","sequence":"first","affiliation":[{"name":"Department of Orthopaedic SurgeryLeiden University Medical CenterLeidenThe Netherlands"},{"name":"Center for Bone QualityLeiden University Medical CenterLeidenThe Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natasha M","family":"Appelman-Dijkstra","sequence":"additional","affiliation":[{"name":"Department of MedicineDivision of EndocrinologyLeiden University Medical CenterLeidenThe Netherlands"},{"name":"Center for Bone QualityLeiden University Medical CenterLeidenThe Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martha","family":"Fiocco","sequence":"additional","affiliation":[{"name":"Mathematical InstituteLeiden UniversityLeidenThe Netherlands"},{"name":"Department of Medical Statistics and BioinformaticsLeiden University Medical CenterLeidenThe Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michiel AJ","family":"van de Sande","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic SurgeryLeiden University Medical CenterLeidenThe Netherlands"},{"name":"Center for Bone QualityLeiden University Medical CenterLeidenThe Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"PD Sander","family":"Dijkstra","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic SurgeryLeiden University Medical CenterLeidenThe Netherlands"},{"name":"Center for Bone QualityLeiden University Medical CenterLeidenThe Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neveen AT","family":"Hamdy","sequence":"additional","affiliation":[{"name":"Department of MedicineDivision of EndocrinologyLeiden University Medical CenterLeidenThe Netherlands"},{"name":"Center for Bone QualityLeiden University Medical CenterLeidenThe Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2016,9,20]]},"reference":[{"key":"2024021014254054400_jbmr2999-bib-0001","first-page":"777","article-title":"Fibrous dysplasia of bone: a condition affecting one, several or many bones, graver cases of which may present abnormal pigmentation of skin, premature sexual development, hyperthyroidism or still other 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