Long-term effect of growth hormone on sleep-disordered breathing in Malaysian children with Prader-Willi syndrome: a retrospective study

J Clin Sleep Med. 2024 Aug 1;20(8):1291-1299. doi: 10.5664/jcsm.11140.

Abstract

Study objectives: The effect of recombinant human growth hormone (rhGH) on sleep-disordered breathing (SDB) in Malaysian children with Prader-Willi syndrome (PWS) is under-investigated. We determined (1) the short- and long-term effects of rhGH and (2) factors associated with worsening SDB in children with PWS receiving rhGH.

Methods: This retrospective study included children with PWS (with and without rhGH) who had undergone at least 1 polysomnography. Outcomes measured were the presence of SDB before and after starting rhGH and the progress of SDB with and without rhGH. Serial insulin-like growth factor 1 (IGF-1) measurements were recorded.

Results: One-hundred and thirteen polysomnograms were analyzed. The majority (92.3%) of initial polysomnograms showed SDB, with a median (interquartile range) apnea-hypopnea index of 5.0 (2.6, 16.3) events/h. The age for receiving rhGH was a median (IQR) of 1.9 (0.7, 3.4) years. One-third (36.8%) had worsening SDB after initiating rhGH, which was associated with higher IGF-1 levels post-rhGH (P = .007). After a median of 5 years of rhGH, 73.6% maintained or reduced their positive airway pressure settings. Without rhGH, 80% had increased their positive airway pressure settings. Worsening SDB while on rhGH was associated with higher body mass index, lower rhGH dose, higher IGF-1 levels, and non-15q deletion.

Conclusions: The majority of Malaysian children with PWS had SDB. At initiation of rhGH, one-third of patients had worsening SDB, associated with increased IGF-1 levels. Stabilization of SDB was more frequently seen in those receiving long-term rhGH. Worsening SDB while on rhGH was associated with a higher body mass index, receiving a lower dose of rhGH, higher IGF-1 levels, and non-15q deletion.

Citation: Tan YT, Azanan MS, Hng SY, et al. Long-term effect of growth hormone on sleep-disordered breathing in Malaysian children with Prader-Willi syndrome: a retrospective study. J Clin Sleep Med. 2024;20(8):1291-1299.

Keywords: Malaysia; Prader Willi syndrome; central sleep apnea; growth hormone; obstructive sleep apnea; polysomnography; sleep-disordered breathing.

MeSH terms

  • Child
  • Child, Preschool
  • Female
  • Human Growth Hormone* / therapeutic use
  • Humans
  • Infant
  • Insulin-Like Growth Factor I
  • Malaysia / epidemiology
  • Male
  • Polysomnography*
  • Prader-Willi Syndrome* / complications
  • Prader-Willi Syndrome* / drug therapy
  • Retrospective Studies
  • Sleep Apnea Syndromes* / drug therapy
  • Treatment Outcome

Substances

  • Human Growth Hormone
  • Insulin-Like Growth Factor I