A 24-Week Physical Activity Intervention Increases Bone Mineral Content without Changes in Bone Markers in Youth with PWS

Abstract

Bone mineral density (BMD) is of concern in Prader-Willi syndrome (PWS). This study compared responses to a physical activity intervention in bone parameters and remodeling markers in youth with PWS (n = 45) and youth with non-syndromic obesity (NSO; n = 66). Measurements occurred at baseline (PRE) and after 24 weeks (POST) of a home-based active games intervention with strengthening and jumping exercises (intervention group = I) or after a no-intervention period (control group = C). Dual x-ray absorptiometry scans of the hip and lumbar spine (L1-L4) determined BMD and bone mineral content (BMC). Bone markers included fasting bone-specific alkaline phosphatase (BAP) and C-terminal telopeptide of type I collagen (CTx). Both I and C groups increased their hip BMD and BMC (p < 0.001). Youth with PWS-I increased their spine BMC from PRE to POST (p < 0.001) but not youth with PWS-C (p = 1.000). Youth with NSO (I and C) increased their spine BMC between PRE and POST (all p < 0.001). Youth with PWS showed lower BAP (108.28 ± 9.19 vs. 139.07 ± 6.41 U/L; p = 0.006) and similar CTx (2.07 ± 0.11 vs.1.84 ± 0.14 ng/dL; p = 0.193) than those with NSO regardless of time. Likely, the novelty of the intervention exercises for those with PWS contributed to gains in spine BMC beyond growth. Bone remodeling markers were unaltered by the intervention.

Physical Activity in Patients with Prader-Willi Syndrome—A Systematic Review of Observational and Interventional Studies

“…In these intervention studies, eight distinct PA interventions were assessed in one randomized controlled trial (RCT) [ 14 ], four non-randomized controlled trials [ 19 , 20 , 21 , 22 ] and three single-group intervention studies [ 8 , 23 , 24 ]. Six additional studies reported the effect of these eight interventions [ 13 , 15 , 16 , 25 , 26 , 27 ]. Among the five randomized or non-randomized controlled trials, two were rated as good-quality trials [ 14 , 21 ], two as a fair-quality trial [ 19 , 20 ], and two as poor-quality trials [ 22 ].…”

“…This is in agreement with the known effect of PA in adults with non-syndromic obesity, in whom only a weight loss of small magnitude (2 to 3 kg on average) is observed after an exercise training program [ 37 ]. In children with PWS, PA interventions seem to have a beneficial effect on lean body mass [ 20 , 22 , 26 ] and bone mineral density, which is an important major health benefit in these patients with low baseline values of lean body mass and bone mineral density [ 15 ]. Observational studies have also reported a positive relation between habitual PA and lean body mass [ 22 , 29 ] or bone parameters [ 11 ].…”

“…Observational studies have also reported a positive relation between habitual PA and lean body mass [ 22 , 29 ] or bone parameters [ 11 ]. Importantly, a large majority of children and adults participating in the PA intervention were on GH replacement therapy at the time of the study or had been on such therapy for extended periods of time before the study [ 15 , 20 , 22 , 26 ]. In this context, PA is likely to reinforce the effects of GH therapy, and it has therefore been described as a co-adjuvant intervention to GH therapy in children with PWS [ 3 ].…”

“…Given the relatively few studies included and their diversity in terms of population, interventions, and outcomes assessed, this review did not provide a quantitative analysis of findings. Since then, several studies were published [ 8 , 13 , 14 , 15 , 16 ], adding to the body of evidence on the effect of PA interventions in patients with PWS. Importantly, some of these studies described in detail the implementation process of the PA intervention [ 8 , 13 , 16 ], which is of great importance to facilitate their transferability in real-life settings.…”

Special Issue: Genetics of Prader–Willi Syndrome

“…They propose that further longitudinal studies are needed to characterize the gut microbiota profile in PWS and its role. Rubin et al [ 13 ] then reported on a 24-week physical activity intervention program in PWS and found increases in bone mineral content without changes in bone markers in the youth with PWS via this intervention. They found that the youth with PWS had increased spine bone mineral content following physical activity interventions; however, bone remodeling markers remained unaltered.…”