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Physical training and high-protein diet improved muscle strength, parent-reported fatigue, and physical quality of life in children with Pompe disease.

Journal of inherited metabolic disease
July 1, 2023
L E Scheffers et al. (9 authors)
Randomized Controlled TrialJournal ArticleResearch Support, Non-U.S. Gov'tHuman StudyClinical
Study Details

Study Goal

The researchers aimed to investigate the effects of a 12-week tailored lifestyle intervention, including a high-protein diet (2 grams/kg), on exercise capacity and other health outcomes in children with Pompe disease.

Results Summary

The intervention improved muscle strength, core stability, quality of life, and parent-reported fatigue, with significant increases in absolute Peak VO2 and specific muscle groups' strength. However, improvements in exercise capacity were not significant compared to the control period.

Population

Children with Pompe disease (median age 10.6 years, including six classic infantile patients).

Effective Dosage

2 grams/kg of body weight.

Duration

12 weeks.

Interactions

None mentioned.

Extracted Claims (12)
InterventionDirectionEndpointPopulationDosageImpactClaim #
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
no change
safety
children with Pompe disease
-
seemed safe
#1
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
absolute Peak VO2
children with Pompe disease
1279 mL/min [1012.5-2006] vs. 1352 mL/min [1101.5-2069]
improved significantly
#2
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
Muscle strength of the hip flexors, hip abductors, elbow extensors, neck extensors, knee extensors
children with Pompe disease
-
improved significantly
#3
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
core stability
children with Pompe disease
-
improved significantly
#4
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
the change in health domain of quality of life
children with Pompe disease
-
reported a significant increase
#5
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
quality of life domains: physical functioning, change in health, family cohesion
children with Pompe disease
-
reported significantly better scores
#6
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
decrease
fatigue
children with Pompe disease
-
reported significantly better scores
#7
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
muscle strength
children with Pompe disease
-
led to improvements
#8
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
core stability
children with Pompe disease
-
led to improvements
#9
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
quality of life
children with Pompe disease
-
led to improvements
#10
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
decrease
parent-reported fatigue
children with Pompe disease
-
led to improvements
#11
a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg)
increase
disease trajectory
Pompe patients with a stable disease trajectory
-
seemed to benefit the most
#12
Abstract

Exercise has proven to be an effective adjuvant treatment to enzyme replacement therapy (ERT) in mildly affected adult Pompe patients. The aim of this study was to investigate the effects of a 12-week tailored lifestyle intervention, consisting of physical training and a high protein diet (2 grams/kg), in children with Pompe disease. This randomized controlled semi-crossover trial investigated the effects of a lifestyle intervention on the primary outcome: exercise capacity. Secondary outcomes were: muscle strength, core stability, motor function, physical activity levels, quality of life, fatigue, fear of exercise, caloric intake, energy balance, body composition, and safety. Fourteen Pompe patients with a median age of 10.6 [IQR: 7.2-14.5], of whom six classic infantile patients, participated in the lifestyle intervention. At baseline, patients had a lower exercise capacity compared to healthy peers (median 70.3% [IQR: 54.8%-98.6%] of predicted). After the intervention, absolute Peak VO2 improved significantly (1279 mL/min [1012.5-2006] vs. 1352 mL/min [1101.5-2069], p = 0.039), but not compared to the control period. Muscle strength of the hip flexors, hip abductors, elbow extensors, neck extensors, knee extensors, and core stability improved significantly compared to the control period. Children reported a significant increase on the change in health domain of quality of life, parents reported significantly better scores on the quality of life domains: physical functioning, change in health, family cohesion, and fatigue. A 12-week tailored lifestyle intervention for children with Pompe disease seemed safe and led to improvements in muscle strength, core stability, quality of life, and parent-reported fatigue. Pompe patients with a stable disease trajectory seemed to benefit the most from the intervention.

Medical Subject Headings (MeSH)
ChildHumansDiet, High-ProteinExerciseFatigueGlycogen Storage Disease Type IIMuscle StrengthQuality of LifeAdolescent
Study Links
Quality Scores
Safety85
Efficacy75/10
Quality80/10
Citation Metrics
Total Citations6
Citations/Year3.0
Relative Citation Ratio1.89
NIH Percentile72.9%
Research Impact Scores
APT Score0.50
Weight Score2.72
Normalized Score0.80
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