Association between physical activity and bone in children with Prader-Willi syndrome

Duran, Andrea T.; Wilson, Kathleen; Castner, Diobel M.; Tucker, Jared M.; Rubin, Daniela A.

doi:10.1515/jpem-2015-0233

Cited by 13 publications

(16 citation statements)

References 46 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Objectively measured habitual PA was found to be positively associated with lean body mass in children and adults with PWS [ 22 , 29 ] and with bone parameters (hip and body bone mineral content and bone mineral density) in children with PWS [ 11 ]. No significant association was reported with body fat in either children or adults with PWS [ 7 , 10 , 31 , 32 ].…”

Section: Resultsmentioning

confidence: 99%

“…However, less than 10% of children with PWS reach the recommended level of PA [ 6 , 7 ], and this proportion does not exceed 20% in adults [ 8 , 9 ]. Over the last 10 years, a number of studies have assessed habitual PA and, to a lesser extent, sedentary behavior in these patients [ 7 , 8 , 9 , 10 , 11 ]. Most studies used objective methods such as accelerometers that are considered as the method of reference for measuring PA and sedentary time, especially in patients with cognitive impairment for whom self-reporting may be particularly challenging [ 12 ].…”

Section: Introductionmentioning

confidence: 99%

See 1 more Smart Citation

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

Bellicha

Coupaye

Mosbah

et al. 2021

JCM

View full text Add to dashboard Cite

Physical activity (PA) is an important aspect of the management of patients with Prader-Willi syndrome (PWS). However, the day-to-day implementation of PA programs is particularly challenging in these patients. This systematic review aimed (1) to describe habitual PA and sedentary behavior and (2) to assess the effects of PA interventions and to describe their implementation process, in children and adults with PWS. A systematic search of controlled trials, single-group interventions, observational, and qualitative studies published up to December 2020 was performed. Twenty-five studies were included. Habitual PA was found to be lower in patients with PWS compared to controls without obesity or with non-syndromic obesity. Habitual PA was positively associated with lean body mass and bone parameters in children with PWS, and these finding were strengthened by intervention studies reporting an increase in both outcomes after a PA program. PA programs also improved physical function (muscle strength, walking distance, and coordination), without significant effect on weight and fat mass. Attendance to exercise sessions was usually high and no serious adverse effect was reported. In conclusion, supervised PA programs are beneficial for children and adults with PWS. Support should be provided to families to facilitate their implementation in real-life settings.

show abstract

Section: Resultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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

Bellicha

Coupaye

Mosbah

et al. 2021

JCM

View full text Add to dashboard Cite

show abstract

“…Butler et al found low bone mass or osteopenia in four of the nine subjects with PWS over the age of 20 years 4. Past reports have described that growth hormone and gonadal hormone treatment is effective for elevating BMD in young PWS patients 6,13. However, a recent report suggested that GH treatment is ineffective in adult PWS patients 14.…”

Section: Discussionmentioning

confidence: 99%

<p>Efficacy of denosumab therapy for a 21-year-old woman with Prader-Willi syndrome, osteoporosis and history of fractures: a case report</p>

Uehara

Nakamura

Takahashi

et al. 2019

TCRM

View full text Add to dashboard Cite

Appropriate management for osteoporosis in adult patients with Prader-Willi syndrome (PWS) has not been established. We report on a 21-year-old woman with PWS, who underwent denosumab treatment for osteoporosis. She presented with fractures and was shown to have very low bone mineral density (BMD), while she had been treated with supplementation of growth hormone for 7–14 years of age and estrogen from 15 years of age. BMD was monitored in the total hip region by dual-energy X-ray absorptiometry. Laboratory tests included bone-specific alkaline phosphatase, urinary type I collagen amino-terminal telopeptide, tartrate-resistant acid phosphatase 5b, 1-alpha, 25-dihydroxyvitamin D3, and parathyroid hormone. BMD and laboratory data were evaluated before and at 4, 8, and 13 months of treatment. After 13 months of denosumab therapy, BMD increased by 4.5%, and bone turnover markers notably improved. No fractures occurred. To the best of our knowledge, this is the first report to describe the clinical outcomes of denosumab treatment for osteoporosis in patients with PWS. Based on our findings, denosumab could represent an effective treatment option for osteoporosis in PWS patients.

show abstract

“…In a recent study in youth with PWS who had been on GHRT for at least two years, we showed that youth with PWS showed lower BMD at the hips than height-matched controls with obesity [ 4 ]. This finding is potentially due to insufficient ambulatory activity and stimulus for bone formation [ 5 ]. Weight-bearing exercise applies mechanical forces on the bones through the ground reaction forces and the contractile activity of the muscles [ 6 ].…”

Section: Introductionmentioning

confidence: 99%

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

Rubin

Wilson

Orsso

et al. 2020

Genes

Self Cite

View full text Add to dashboard Cite

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.

show abstract

Association between physical activity and bone in children with Prader-Willi syndrome

Abstract: PA of at least moderate intensity appears important for BMC and BMD in children with PWS.

Cited by 13 publications

References 46 publications

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

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

<p>Efficacy of denosumab therapy for a 21-year-old woman with Prader-Willi syndrome, osteoporosis and history of fractures: a case report</p>

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

Contact Info

Product

Resources

About