{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T18:51:19Z","timestamp":1791312679218,"version":"4.3.1"},"reference-count":74,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T00:00:00Z","timestamp":1791244800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T00:00:00Z","timestamp":1791244800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Endocrinology"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Context<\/jats:title>\n                    <jats:p>Hypogonadism in Prader\u2010Willi syndrome (PWS) is associated with decreased bone mineral density (BMD) in male adolescents and adults. Testosterone Replacement Therapy (TRT) improves BMD and sexual maturation, but TRT could aggravate pre\u2010existing challenging behaviour in male patients with PWS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To investigate the effect of TRT on behaviour, BMD and body composition in adolescents and young adults with PWS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design<\/jats:title>\n                    <jats:p>Longitudinal observational study in a Dutch PWS cohort.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Participants<\/jats:title>\n                    <jats:p>Fifty\u2010nine male patients with PWS on growth hormone treatment, aged &gt;\u200914 years.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      Behaviour was assessed by the Developmental Behaviour Checklist\u2010Parents(DBC\u2010P). Dual energy X\u2010ray absorptiometry scans were performed, measuring BMD of the lumbar spine (BMD\n                      <jats:sub>LS<\/jats:sub>\n                      ) and total body (BMD\n                      <jats:sub>TB<\/jats:sub>\n                      ), lean body mass (LBM) and fat mass percentage (FM%).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      59 patients with a median (IQR) age of 22.57 (18.46\u201326.35) years, of which 44 used TRT. Median age at TRT initiation was 16.6 years. Three patients (6.8%) discontinued TRT permanently due to challenging behaviour. In 30 patients (68.2%), there was no behaviour necessitating a dose change or discontinuation. DBC\u2010P total score did not significantly change during TRT. BMD SDS stabilised following TRT initiation, with a mean (95% CI) BMD SDS difference from baseline of 0.24 (\u22120.06 to 0.54;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.107) BMD\n                      <jats:sub>LS<\/jats:sub>\n                      and \u22120.28 (\u22120.50 to \u22120.06;\n                      <jats:italic>p<\/jats:italic>\n                      \u2009=\u20090.014) BMD\n                      <jats:sub>TB<\/jats:sub>\n                      after 5 years. LBM SDS improved and FM% SDS remained similar.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Under close surveillance, TRT is an efficacious method to maintain BMD and improve body composition in hypogonadal male patients with PWS, with minor effects on behaviour in most cases.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/cen.70213","type":"journal-article","created":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T11:02:32Z","timestamp":1791284552000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Testosterone Replacement Therapy in Adolescents and Young Adults With Prader\u2010Willi Syndrome: Efficacy and Effects on Behaviour"],"prefix":"10.1111","author":[{"ORCID":"https:\/\/orcid.org\/0009-0001-4515-0702","authenticated-orcid":false,"given":"Wiebe R.","family":"Flamman","sequence":"first","affiliation":[{"name":"Department of Pediatrics Subdivision of Endocrinology, Erasmus University Medical Center\u2010Sophia Children's Hospital Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6882-0086","authenticated-orcid":false,"given":"Demi J.","family":"Trueba\u2010Timmermans","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Subdivision of Endocrinology, Erasmus University Medical Center\u2010Sophia Children's Hospital Rotterdam The Netherlands"},{"name":"Dutch Reference Center for Prader\u2010Willi Syndrome Rotterdam The Netherlands"},{"name":"Dutch Growth Research Foundation Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lionne N.","family":"Grootjen","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Subdivision of Endocrinology, Erasmus University Medical Center\u2010Sophia Children's Hospital Rotterdam The Netherlands"},{"name":"Dutch Reference Center for Prader\u2010Willi Syndrome Rotterdam The Netherlands"},{"name":"Dutch Growth Research Foundation Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura C. G.","family":"de Graaff","sequence":"additional","affiliation":[{"name":"Dutch Reference Center for Prader\u2010Willi Syndrome Rotterdam The Netherlands"},{"name":"Department of Internal Medicine Division of Endocrinology, Erasmus Medical Center, University Medical Centre Rotterdam Rotterdam The Netherlands"},{"name":"Department of Internal Medicine Division of Endocrinology, Center for Adults with Rare Genetic Syndromes, Erasmus Medical Center, University Medical Center Rotterdam Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joyce M.","family":"Geelen","sequence":"additional","affiliation":[{"name":"Dutch Reference Center for Prader\u2010Willi Syndrome Rotterdam The Netherlands"},{"name":"Department of Pediatrics, Subdivision of Genetic and Developmental Pediatrics Radboud University Medical Center, Amalia Children's Hospital Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janielle A. E. 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