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International Journal of Pediatric Endocrinology
Published in: International Journal of Pediatric Endocrinology 1/2018

Open Access 01-12-2018 | Research

Body composition, adipokines, bone mineral density and bone remodeling markers in relation to IGF-1 levels in adults with Prader-Willi syndrome

Authors: I. Caroline van Nieuwpoort, Jos W. R. Twisk, Leopold M. G. Curfs, Paul Lips, Madeleine L. Drent

Published in: International Journal of Pediatric Endocrinology | Issue 1/2018

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Abstract

Background

In patients with Prader-Willi syndrome (PWS) body composition is abnormal and alterations in appetite regulating factors, bone mineral density and insulin-like growth factor-1 (IGF-1) levels have been described. Studies in PWS adults are limited. In this study, we investigated body composition, appetite regulating peptides, bone mineral density and markers of bone remodeling in an adult PWS population. Furthermore, we investigated the association between these different parameters and IGF-1 levels because of the described similarities with growth hormone deficient patients.

Methods

In this cross-sectional observational cohort study in a university hospital setting we studied fifteen adult PWS patients. Anthropometric and metabolic parameters, IGF-1 levels, bone mineral density and bone metabolism were evaluated. The homeostasis model assessment of insulin resistance (HOMA2-IR) was calculated. Fourteen healthy siblings served as a control group for part of the measurements.

Results

In the adult PWS patients, height, fat free mass, IGF-1 and bone mineral content were significantly lower when compared to controls; body mass index (BMI), waist, waist-to-hip ratio and fat mass were higher. There was a high prevalence of osteopenia and osteoporosis in the PWS patients. Also, appetite regulating peptides and bone remodelling markers were aberrant when compared to reference values. Measurements of body composition were significantly correlated to appetite regulating peptides and high-sensitive C-reactive protein (hs-CRP), furthermore HOMA was correlated to BMI and adipokines.

Conclusion

In adults with Prader-Willi syndrome alterations in body composition, adipokines, hs-CRP and bone mineral density were demonstrated but these were not associated with IGF-1 levels. Further investigations are warranted to gain more insight into the exact pathophysiology and the role of these alterations in the metabolic and cardiovascular complications seen in PWS, so these complications can be prevented or treated as early as possible.
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Metadata
Title
Body composition, adipokines, bone mineral density and bone remodeling markers in relation to IGF-1 levels in adults with Prader-Willi syndrome
Authors
I. Caroline van Nieuwpoort
Jos W. R. Twisk
Leopold M. G. Curfs
Paul Lips
Madeleine L. Drent
Publication date
01-12-2018
Publisher
BioMed Central
Published in
International Journal of Pediatric Endocrinology / Issue 1/2018
Electronic ISSN: 1687-9856
DOI
https://doi.org/10.1186/s13633-018-0055-4

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