Effects of 8 years of growth hormone treatment on scoliosis in children with Prader–Willi syndrome

in European Journal of Endocrinology
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  • 1 Dutch Reference Center for Prader-Willi Syndrome, Rotterdam, The Netherlands
  • 2 Department of Pediatrics, Subdivision of Endocrinology, Erasmus University Medical Center-Sophia Children’s Hospital, Rotterdam, The Netherlands
  • 3 Department of Orthopaedic Surgery, Erasmus University Medical Center, Rotterdam, The Netherlands
  • 4 Dutch Growth Research Foundation, Rotterdam, The Netherlands

Correspondence should be addressed to L N Grootjen Email l.grootjen@kindengroei.nl
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Objective

Scoliosis is frequently seen in children with Prader–Willi syndrome (PWS). There is still concern that growth hormone (GH) treatment might increase the risk of onset or progression of scoliosis. Short-term data suggested no adverse effects of GH on scoliosis, but long-term effects of GH treatment on development of scoliosis in PWS are unknown. This study investigated the effects of 8 years of GH treatment on scoliosis in children with PWS.

Design

Open-label, prospective cohort study in 103 children with PWS receiving GH for 8 years was analyzed. Prevalence and severity of scoliosis were compared to a group of 23 age-matched GH-untreated children with PWS.

Methods

Spine X-rays and DEXA-scans were performed, and Cobb angel was measured by two independent observers.

Results

After 8 years of GH treatment, at median age of 10.8 years, prevalence of scoliosis was 77.7%. No difference in prevalence or severity of scoliosis was found between GH-treated and age-matched untreated children with PWS (P = 0.409 and P = 0.709, respectively). Height SDS and trunkLBM were significantly higher in GH-treated children. Higher bone mineral density of the lumbar spine was found in children without scoliosis after 8 years of GH. Bone mineral apparent density of lumbar spine (BMADLS) SDS was associated with lower Cobb angle (r = −0.270, P = 0.008).

Conclusions

Eight years of GH treatment has no adverse effects on the prevalence and severity of scoliosis in children with PWS until 11 years of age. As BMADLS SDS is inversely associated with Cobb angle, it is pivotal to optimize the BMD status in children with PWS.

 

     European Society of Endocrinology

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