Sleep-Related Breathing Disorders in Young Adults with Prader-Willi Syndrome: A Placebo-Controlled, Crossover GH Trial
Context: Sleep-related breathing disorders (SRBD) are common in people with Prader-Willi syndrome (PWS). Young adults with PWS benefit from GH continuation after attaining adult height by maintaining the improved body composition obtained during childhood. There are, no studies about the effects of GH on SRBD in young adults with PWS who were treated with GH during childhood. Objective: Investigate the effects of GH vs placebo on SRBD in young adults with PWS who were treated with GH during childhood and had attained adult height. Design: Two-year, randomized, double-blind, placebo-controlled, crossover study in 27 young adults with PWS, stratified for sex and body mass index. Setting: Dutch PWS Reference Center. Intervention: Crossover intervention with GH (0.67 mg/m2/d) and placebo, both over one year. Main Outcome Measures: Apnea hypopnea index (AHI), obstructive apnea index (OAI), central apnea index (CAI), measured by polysomnography. Results: Compared with placebo, GH did not increase AHI, CAI, or OAI (P > 0.35). The effect of GH vs placebo was neither different between men and women, nor between patients with a deletion or maternal uniparental disomy/imprinting center defect. After two years, there was no difference in AHI, CAI, or OAI compared with baseline (P > 0.18). Two patients (7%) fulfilled the criteria of obstructive sleep apnea regardless of GH or placebo. Conclusions: GH compared with placebo does not cause a substantial increase in AHI, CAI, or OAI in adults with PWS who were treated with GH during childhood and have attained adult height. Our findings are reassuring and prove that GH can be administered safely.
|Persistent URL||dx.doi.org/10.1210/jc.2019-00391, hdl.handle.net/1765/127464|
|Journal||Journal of Clinical Endocrinology and Metabolism|
Donze, S.H, de Weerd, A.W, van den Bossche, R.A, Joosten, K.F.M, & Hokken-Koelega, A.C.S. (2019). Sleep-Related Breathing Disorders in Young Adults with Prader-Willi Syndrome: A Placebo-Controlled, Crossover GH Trial. Journal of Clinical Endocrinology and Metabolism, 104(9), 3931–3938. doi:10.1210/jc.2019-00391