{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,14]],"date-time":"2026-10-14T10:34:22Z","timestamp":1791974062375,"version":"4.4.0"},"reference-count":28,"publisher":"Oxford University Press (OUP)","issue":"7","license":[{"start":{"date-parts":[[2023,5,21]],"date-time":"2023-05-21T00:00:00Z","timestamp":1684627200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100002429","name":"Amgen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002429","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100005040","name":"Bontius Stichting","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100005040","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100013220","name":"Ultragenyx Pharmaceutical","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100013220","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["asbmr.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["J of Bone &amp; Mineral Res"],"published-print":{"date-parts":[[2023,7]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    This study aimed to evaluate the prevalence of and risk factors for coxa vara deformity in patients with fibrous dysplasia\/McCune\u2010Albright syndrome (FD\/MAS). This study was conducted at the National Institutes of Health and Leiden University Medical Center. All patients with any subtype of FD\/MAS, FD involving the proximal femur, one or more X\u2010rays available and age\u2009&lt;30\u2009years were included. X\u2010rays were scored for the neck\u2010shaft angle (NSA). Varus deformity was defined as NSA &lt;110 degrees or\u2009&gt;10 degrees below age\u2010specific values. Risk factors for deformity were assessed by nested case\u2013control analysis, comparing patients and femurs with and without deformity, and by linear mixed effects model, modeling temporal NSA decrease (the natural course of the NSA) in non\u2010operated femurs with two or more X\u2010rays. Assessed variables included growth hormone excess, hyperthyroidism, hypophosphatemia, &gt;25% of the femur affected, calcar destruction, radiolucency, and bilateral involvement. In total 180 patients were studied, 57% female. Mean \u00b1 SD baseline age was 13.6 \u00b1\u20097.5\u2009years; median follow\u2010up 5.4 (interquartile range [IQR], 11.1) years. Sixty\u2010three percent (63%) were diagnosed with MAS. A total of 94 patients were affected bilaterally; 274 FD femurs were analyzed; 99 femurs had a varus deformity (36%). In the nested case\u2013control analysis, risk factors were as follows: presence of MAS (\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.001), hyperthyroidism (\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.001), hypophosphatemia (\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.001), high percentage of femur affected (\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.001), and calcar destruction (\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.001). The linear mixed effects model included 114 femurs, identified risk factors were: growth hormone excess (\u03b2\u2009=\u20097.2,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009=\u20090.013), hyperthyroidism (\u03b2\u2009=\u200911.3,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009&lt;\u20090.001), &gt;25% of the femur affected (\u03b2\u2009=\u200913.2,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009=\u20090.046), calcar destruction (\u03b2\u2009=\u20098.3,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009=\u20090.004), radiolucency (\u03b2\u2009=\u20093.9,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009=\u20090.009), and bilateral involvement (\u03b2\u2009=\u20099.8,\n                    <jats:italic>p<\/jats:italic>\n                    \u2009=\u20090.010). Visual inspection of the graph of the model demonstrated most progression of deformity if NSA &lt;120 degrees with age\u2009&lt;\u200915\u2009years. In conclusion, in tertiary care centers, the prevalence of FD\/MAS coxa vara deformity was 36%. Risk factors included presence of MAS, high percentage of femur affected, calcar destruction, radiolucency, NSA &lt;120 degrees and age\u2009&lt;\u200915\u2009years. \u00a9 2023 The Authors.\n                    <jats:italic>Journal of Bone and Mineral Research<\/jats:italic>\n                    published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research (ASBMR).\n                  <\/jats:p>","DOI":"10.1002\/jbmr.4818","type":"journal-article","created":{"date-parts":[[2023,4,27]],"date-time":"2023-04-27T07:39:17Z","timestamp":1682581157000},"page":"968-975","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":9,"title":["Coxa Vara Deformity in Fibrous Dysplasia\/\n                    <scp>McCune\u2010Albright<\/scp>\n                    Syndrome: Prevalence, Natural History and Risk Factors: A Two\u2010Center Study"],"prefix":"10.1093","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6258-2531","authenticated-orcid":false,"given":"Maartje E.","family":"Meier","sequence":"first","affiliation":[{"name":"Department of Orthopaedic Surgery, Center for Bone Quality Leiden University Medical Center  Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5035-127X","authenticated-orcid":false,"given":"Natasha M.","family":"Appelman\u2010Dijkstra","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Endocrinology, Center for Bone Quality Leiden University Medical Center  Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3676-7644","authenticated-orcid":false,"given":"Michael T.","family":"Collins","sequence":"additional","affiliation":[{"name":"Skeletal Disorders &amp; Mineral Homeostasis Section, National Institute of Dental and Craniofacial Research National Institutes of Health  Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Raya E.S.","family":"Geels","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Endocrinology, Center for Bone Quality Leiden University Medical Center  Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Robert P.","family":"Stanton","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery Nemours Children's Hospital  Orlando FL USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pieter Bas","family":"de Witte","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Center for Bone Quality Leiden University Medical Center  Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alison M.","family":"Boyce","sequence":"additional","affiliation":[{"name":"Metabolic Bone Disorders Unit, National Institute of Dental and Craniofacial Research National Institutes of Health  Bethesda MD USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michiel A.J.","family":"van de Sande","sequence":"additional","affiliation":[{"name":"Department of Orthopaedic Surgery, Center for Bone Quality Leiden University Medical Center  Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2023,5,21]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199112123252403"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-019-1102-9"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0000000000000473"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1302\/0301-620X.96B5.33281"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13018-020-02073-y"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.2106\/JBJS.N.00230"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-7-S1-S1"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.5312\/wjo.v13.i3.329"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1097\/BPO.0000000000002097"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-018-0805-7"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/jbmr.4463"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11999-016-4806-3"},{"key":"e_1_2_8_14_1","first-page":"73","article-title":"Coxa vara in children","volume":"22","author":"Amstutz HC","year":"1962","journal-title":"Clin Orthop Relat Res"},{"key":"e_1_2_8_15_1","first-page":"376","volume-title":"Coxa Vara in Developmental and Acquired Abnormalities of the Femur","author":"Shapiro F","year":"2001"},{"key":"e_1_2_8_16_1","first-page":"765","volume-title":"Tachdjian's Pediatric Orthopaedics","author":"Herring J","year":"2002"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11999-010-1447-9"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1097\/01241398-200011000-00007"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1259\/bjr\/29690721"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11999-013-3380-1"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13018-015-0313-6"},{"issue":"4","key":"e_1_2_8_22_1","first-page":"304","article-title":"Coxa vara: another option for fixation","volume":"10","author":"Burns 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