This Article Full Text Full Text (PDF) Submit a related Letter to the Editor Alert me when this article is cited Alert me when eLetters are posted Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Download to citation manager Request Copyright Permission Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by de Lind van Wijngaarden, R. F. A. Articles by Hokken-Koelega, A. C. S. Search for Related Content PubMed PubMed Citation Articles by de Lind van Wijngaarden, R. F. A. Articles by Hokken-Koelega, A. C. S. Related Collections Pediatric Endocrinology

Randomized Controlled Trial to Investigate the Effects of Growth Hormone Treatment on Scoliosis in Children with Prader-Willi Syndrome

Dutch Growth Research Foundation (R.F.A.d.L.v.W., D.A.M.F., A.C.S.H.-K.), 3016 AH Rotterdam, The Netherlands; Departments of Pediatric Endocrinology (R.F.A.d.L.v.W., D.A.M.F., A.C.S.H.-K.), and Pediatric Orthopedic Surgery (L.W.L.d.K.), Erasmus University Medical Center/Sophia Children’s Hospital, 3015 GJ Rotterdam, The Netherlands; The Netherlands Institute for Health Sciences (H.J.D.), Erasmus University Rotterdam, 3015 GE Rotterdam, The Netherlands; and Department of Pediatric Endocrinology (B.J.O.), University Medical Center St. Radboud, 6525 GA Nijmegen, The Netherlands

Address all correspondence and requests for reprints to: Roderick de Lind van Wijngaarden, Clinical Research Fellow, Dutch Growth Research Foundation, Erasmus University Medical Center/Sophia Children’s Hospital, Westzeedijk 106, 3016 AH Rotterdam, The Netherlands. E-mail: r.delindvanwijngaarden{at}erasmusmc.nl or r.delind{at}kindengroei.nl.

Context: The prevalence of scoliosis in children with Prader-Willi syndrome (PWS) is 30–80%, depending on age. Although reports about effects of GH treatment on scoliosis in children with PWS are limited, scoliosis is generally considered a contraindication for GH treatment.

Objective: The aim was to study the effects of GH treatment on the onset of scoliosis and curve progression in children with PWS.

Design: We conducted a multicenter, randomized, controlled GH study in infants and prepubertal and pubertal children. Infants and prepubertal children were randomized into a GH-treated group (1.0 mg/m² · d) and a control group for 1 and 2 yr, respectively. Pubertal children were randomized to receive somatropin 1.0 or 1.5 mg/m² · d. Yearly, x-rays of the spine were taken, and height, weight, truncal lean body mass (with dual energy x-ray absorptiometry), and IGF-I were measured.

Patients: A total of 91 children with PWS (median age, 4.7 yr; interquartile range, 2.1–7.4) participated in the study.

Main Outcome Measures: We measured the onset of scoliosis (Cobb >10°) and scoliotic curve progression.

Results: GH-treated children had similar onset of scoliosis and curve progression as randomized controls (P = 0.27–0.79 and P = 0.18–0.98, respectively). GH treatment, IGF-I SD score (SDS), and catch-up growth had no adverse effect on the onset of scoliosis or curve progression, even after adjustment for confounders. Height SDS, truncal lean body mass, and IGF-I SDS were significantly higher in GH-treated children than in randomized controls. At baseline, a higher IGF-I SDS was associated with a lower severity of scoliosis.

Conclusions: Scoliosis should no longer be considered a contraindication for GH treatment in children with PWS.

This article has been cited by other articles:

				R. F. A. de Lind van Wijngaarden, E. P. C. Siemensma, D. A. M. Festen, B. J. Otten, E. G. A. H. van Mil, J. Rotteveel, R. J. H. Odink, G. C. B. Bindels-de Heus, M. van Leeuwen, D. A. J. P. Haring, et al. Efficacy and Safety of Long-Term Continuous Growth Hormone Treatment in Children with Prader-Willi Syndrome J. Clin. Endocrinol. Metab., November 1, 2009; 94(11): 4205 - 4215. [Abstract] [Full Text] [PDF]