Effectiveness of exercise therapy for individuals diagnosed with Charcot–Marie–Tooth disease: A systematic review of randomized clinical trials

• Published in: Journal of the peripheral nervous system 2023-06, Vol.28 (2), p.169-178
• Main Authors: Conde, Rodrigo Melo, Senem, Iara, Santos, Marcia, Lima Osório, Flávia, Marques Júnior, Wilson
• Format: Article
• Language: English
• Subjects: Aerobic capacity; Analgesics; Ankle; Charcot-Marie-Tooth disease; Clinical trials; Exercise; function; Muscle strength; Physical therapy; Quality of life; randomized controlled trial; Systematic review
• ISSN: 1085-9489; 1529-8027
• DOI: 10.1111/jns.12548

Background and Aims Effective treatments for Charcot–Marie–Tooth (CMT) disease lack. Current treatments, such as ankle and foot surgery/orthoses, analgesics, and physiotherapy, focus on relieving the symptoms. Few randomized controlled trials (RCTs) investigated the effectiveness of exercise in patients with CMT, and a systematic review summarizing the effects of such treatments is outdated. This study aims to systematically review the effects of exercise on muscle strength, function, aerobic capacity, and quality of life in CMT. Methods We included RCTs that compared exercise programs against sham exercise, usual care, no exercise, and different exercise programs in individuals diagnosed with CMT. Searches were performed on 10 electronic databases from inception up to July 2021. Authors analyzed titles, s, and full texts and extracted information from the eligible trials. We used the Physiotherapy Evidence Database (PEDro) scale and the GRADE (Grades of Recommendation, Assessment, Development, and Evaluation) approach to evaluate the risk of bias and the certainty of the evidence, respectively. Results were synthesized narratively. Results Eight citations (six studies; pooled n = 214) met the inclusion criteria. The mean age of participants was 38.49 (±13.02) years, and 83% were diagnosed with CMT1A. The mean PEDro score was 5.25 (range 2–9). Six trials were considered to have a high risk of bias. Moderate‐quality evidence suggests that strengthening the ankle dorsiflexors minimizes the progression of weakness at 24 months in children with CMT1A. For other outcomes, quality of the evidence ranged from very low to low. Interpretation Based on the available, evidence we can only recommend exercise to improve muscle strength in children with CMT. More high quality and robust trials are needed.