{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,15]],"date-time":"2026-10-15T16:12:42Z","timestamp":1792080762280,"version":"4.4.0"},"reference-count":24,"publisher":"Oxford University Press (OUP)","issue":"5","content-domain":{"domain":["eje.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2016,11]]},"abstract":"<jats:sec>\n<jats:title>Objective<\/jats:title>\n<jats:p>McCune\u2013Albright syndrome (MAS) is a rare disorder with a broad spectrum including precocious puberty (PP) due to recurrent estrogen-secreting ovarian cysts. This study evaluates the long-term safety and efficacy of letrozole treatment in large cohort of girls with MAS-associated PP.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Design<\/jats:title>\n<jats:p>Retrospective cohort analysis.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Methods<\/jats:title>\n<jats:p>Clinical data, including history and physical examination, bone age, and pelvic ultrasounds, were reviewed on 28 letrozole-treated girls. Adult height was reviewed for 42 historical controls. Outcomes included rate of skeletal maturation, growth velocity, predicted adult height and adult height.<\/jats:p>\n<\/jats:sec>\n<jats:sec>\n<jats:title>Results<\/jats:title>\n<jats:p>Twenty-eight girls received letrozole treatment. Treatment duration was 4.1\u2009\u00b1\u20092.6 years (mean\u2009\u00b1\u20091 <jats:sc>s.d.<\/jats:sc>) (range: 0.5\u201310.9) and mean follow-up was 6.0\u2009\u00b1\u20093.3 years (range: 0.5\u201315.0), for a total of 135.9 person-years of follow-up. Letrozole treatment was highly effective at decreasing the rate of skeletal maturation, with a decline in change in bone age over change in chronological age (\u0394BA\/\u0394CA) from 1.7 (IQR: 2.3) to 0.5 (IQR: 0.4) (<jats:italic>P<\/jats:italic>\u2009&lt;\u20090.0001), and growth velocity <jats:italic>Z<\/jats:italic>-scores, which declined from 2.2\u2009\u00b1\u20092.3 to \u22120.6\u2009\u00b1\u20091.6 (<jats:italic>P<\/jats:italic>\u2009=\u20090.0004). Predicted adult height <jats:italic>Z<\/jats:italic>-scores increased significantly from \u22122.9\u2009\u00b1\u20093.2 to \u22120.8\u2009\u00b1\u20091.5 for subjects on treatment (<jats:italic>P<\/jats:italic>\u2009=\u20090.004). Four subjects who completed treatment reached adult height <jats:italic>Z<\/jats:italic>-scores ranging from \u22121.5 to 1.7 (median: \u22120.6), which were increased in comparison with untreated historical controls (<jats:italic>P<\/jats:italic>\u2009=\u20090.02). There was no change in uterine size or ovarian volumes, and no adverse events over the treatment period.<\/jats:p><\/jats:sec>\n<jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>In this study with the longest follow-up to date, letrozole treatment resulted in sustained beneficial effects on skeletal maturation, growth velocity and predicted adult height.<\/jats:p>\n<\/jats:sec>","DOI":"10.1530\/eje-16-0526","type":"journal-article","created":{"date-parts":[[2016,8,26]],"date-time":"2016-08-26T02:08:56Z","timestamp":1472177336000},"page":"477-483","update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-1","source":"Crossref","is-referenced-by-count":48,"title":["Long-term outcomes of letrozole treatment for precocious puberty in girls with McCune\u2013Albright syndrome"],"prefix":"10.1093","volume":"175","author":[{"given":"Andrea","family":"Estrada","sequence":"first","affiliation":[{"name":"1Section on Skeletal Disorders and Mineral HomestasisCraniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA"},{"name":"2Division of Endocrinology and Diabetes"},{"name":"3Bone Health ProgramDivision of Orthopaedics and Sports Medicine, Children\u2019s National Health System, Washington, District of Columbia, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alison M","family":"Boyce","sequence":"additional","affiliation":[{"name":"1Section on Skeletal Disorders and Mineral HomestasisCraniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA"},{"name":"2Division of Endocrinology and Diabetes"},{"name":"3Bone Health ProgramDivision of Orthopaedics and Sports Medicine, Children\u2019s National Health System, Washington, District of Columbia, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beth A","family":"Brillante","sequence":"additional","affiliation":[{"name":"1Section on Skeletal Disorders and Mineral HomestasisCraniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lori C","family":"Guthrie","sequence":"additional","affiliation":[{"name":"1Section on Skeletal Disorders and Mineral HomestasisCraniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel I","family":"Gafni","sequence":"additional","affiliation":[{"name":"1Section on Skeletal Disorders and Mineral HomestasisCraniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael T","family":"Collins","sequence":"additional","affiliation":[{"name":"1Section on Skeletal Disorders and Mineral HomestasisCraniofacial and Skeletal Diseases Branch, National Institute of Dental and Craniofacial Research, National Institutes of Health, Bethesda, Maryland, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","reference":[{"key":"2016092619334093000_175.5.477.1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM193704292161701"},{"key":"2016092619334093000_175.5.477.2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199112123252403"},{"key":"2016092619334093000_175.5.477.3","unstructured":"Boyce AM Collins MT. 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