Clinical endocrinology · 2026
Testosterone Replacement Therapy in Adolescents and Young Adults With Prader-Willi Syndrome: Efficacy and Effects on Behaviour.
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Flamman Wiebe R, Trueba-Timmermans Demi J, Grootjen Lionne N, de Graaff Laura C G, Geelen Joyce M, van der Velden Janielle A E M, Rings Edmond H H M, Hokken-Koelega Anita C S, Kerkhof Gerthe F
Аннотация
CONTEXT: Hypogonadism in Prader-Willi 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-existing challenging behaviour in male patients with PWS.
OBJECTIVE: To investigate the effect of TRT on behaviour, BMD and body composition in adolescents and young adults with PWS.
DESIGN: Longitudinal observational study in a Dutch PWS cohort.
PARTICIPANTS: Fifty-nine male patients with PWS on growth hormone treatment, aged > 14 years.
METHODS: Behaviour was assessed by the Developmental Behaviour Checklist-Parents(DBC-P). Dual energy X-ray absorptiometry scans were performed, measuring BMD of the lumbar spine (BMDLS) and total body (BMDTB), lean body mass (LBM) and fat mass percentage (FM%).
RESULTS: 59 patients with a median (IQR) age of 22.57 (18.46-26.35) 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-P 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 (-0.06 to 0.54; p = 0.107) BMDLS and -0.28 (-0.50 to -0.06; p = 0.014) BMDTB after 5 years. LBM SDS improved and FM% SDS remained similar.
CONCLUSION: 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.
OBJECTIVE: To investigate the effect of TRT on behaviour, BMD and body composition in adolescents and young adults with PWS.
DESIGN: Longitudinal observational study in a Dutch PWS cohort.
PARTICIPANTS: Fifty-nine male patients with PWS on growth hormone treatment, aged > 14 years.
METHODS: Behaviour was assessed by the Developmental Behaviour Checklist-Parents(DBC-P). Dual energy X-ray absorptiometry scans were performed, measuring BMD of the lumbar spine (BMDLS) and total body (BMDTB), lean body mass (LBM) and fat mass percentage (FM%).
RESULTS: 59 patients with a median (IQR) age of 22.57 (18.46-26.35) 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-P 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 (-0.06 to 0.54; p = 0.107) BMDLS and -0.28 (-0.50 to -0.06; p = 0.014) BMDTB after 5 years. LBM SDS improved and FM% SDS remained similar.
CONCLUSION: 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.