The impact of orthoses on gait in children with Charcot-Marie-Tooth disease

Õunpuu, Sylvia; Garibay, Erin J.; Acsádi, Gyula; Brimacombe, Michael; Pierz, Kristan

doi:10.1016/j.gaitpost.2021.02.005

Cited by 7 publications

(6 citation statements)

References 18 publications

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“…The above is consistent with the increase in ankle power found by Dufek (2014) in a group of adult patients with HMSN thanks to the use of a custom-made carbon fiber brace [18]. Furthermore, our findings confirm that patients tend to undergo a reduction in maximum power at the ankle when using orthoses of greater rigidity [5,35,36]. The only case in which an increase did not occur was the left side of S3.…”

Section: Discussionsupporting

confidence: 90%

Effect of Ankle-Foot Orthoses in Pediatric Patients with Hereditary Motor-Sensory Neuropathy: A Case Series Study

Borghi,

Sassi,

Pandarese

et al. 2023

Children

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(1) Aims: to evaluate the effect on gait performance and standing stability of ankle-foot orthoses (AFO) in pediatric patients with hereditary motor-sensory neuropathy (HMSN). (2) Methods: a retrospective case-series study including three adolescents (S1, S2, S3, mean age 14 years) with HMSN. The subjects were evaluated barefoot, with carbon AFO (Botter) and with solid AFO (SAFO) by means of: gait analysis, stabilometry and gait functional tests (10 Meter Walk Test, 2 Minute Walk Test). Finally, the CSD-OPUS questionnaire was administered to the assess satisfaction and impact of the orthoses on life quality. (3) Results: orthoses improved gait and stability performance. Botter allowed greater ankle movement than SAFO and provided greater push-off power. This, combined with the carbon elastic energy return, might explain better performances in the 2MWT, with a larger distance traveled compared to SAFO for both S1 (110 m vs. 72 m) and S2 (170 m vs. 155 m) and, compared to barefoot walking, also for S3 (211 m vs. 160 m), for which SAFO analysis was not available. Both orthoses improved performance at the stabilometric analysis. The CSD-OPUS questionnaire showed a significantly higher level of satisfaction with Botter for the subjects (S1, S2) who completed the comparison. (4) Conclusions: Both orthoses improved gait and standing, though Botter proved to be better tolerated and more effective in improving gait endurance.

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Section: Discussionsupporting

confidence: 90%

Effect of Ankle-Foot Orthoses in Pediatric Patients with Hereditary Motor-Sensory Neuropathy: A Case Series Study

Borghi,

Sassi,

Pandarese

et al. 2023

Children

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“…The second paper assessed the effect of prescribed ankle-foot orthoses on gait using comprehensive gait analysis techniques. The findings support the consensus statement that ankle-foot orthoses should be used in carefully selected children with impaired functional ambulation, and reinforce that child-specific factors need to be taken account in their prescription 19…”

Section: Resultssupporting

confidence: 79%

Clinical practice guideline for the management of paediatric Charcot-Marie-Tooth disease

Yiu

Bray

Baets

et al. 2022

J Neurol Neurosurg Psychiatry

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Background and objectivesCharcot-Marie-Tooth disease (CMT) is the most common inherited neuropathy and often presents during childhood. Guidelines for the optimal management of common problems experienced by individuals with CMT do not exist, for either children or adults. We formed the Paediatric CMT Best Practice Guidelines Consortium to develop evidence and consensus-based recommendations for the clinical management of children and adolescents with CMT, with the primary objective of promoting optimal, standardised care globally.MethodsDevelopment of this clinical practice guideline involved a series of systematic reviews covering 10 clinical questions, modified Delphi methodology involving an international panel of clinicians to generate consensus where evidence did not exist, and application of the Grading of Recommendations, Assessment, Development and Evaluation (GRADE) approach to evaluate the body of literature and formulate recommendations.ResultsThe final guideline includes three evidence-based and 31 consensus-based recommendations. They encompass the management of muscle weakness, balance and mobility impairment, sensory symptoms, muscle cramps, impaired upper limb function, respiratory impairment, maintenance of joint range of motion and non-surgical management of joint deformity. Consensus was not achieved in some management areas, reflecting differences in practice between clinicians and healthcare settings, and highlighting the need for further research.ConclusionsThis clinical practice guideline provides practical and implementable guidance on the management of common clinical problems experienced by children with CMT and advocates for improved access to multidisciplinary care. Successful dissemination and implementation of these recommendations will be critical in ensuring their application across multiple healthcare settings.

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“…Õunpuu et al 39 examined the impact of orthoses on gait in children with CMT. Their full study cohort showed significant increases in walking velocity when wearing AFOs versus barefoot walking due to a significant increase in stride length 39 . Therefore, assessment of activity and gait in the natural environment may provide a more consistent representation of a participant's function and subsequent progression.…”

Section: Discussionmentioning

confidence: 99%

“…Influential day‐to‐day factors may alter an individual's ability to complete certain assessments ultimately skewing their functional score and disease severity picture. Õunpuu et al 39 examined the impact of orthoses on gait in children with CMT. Their full study cohort showed significant increases in walking velocity when wearing AFOs versus barefoot walking due to a significant increase in stride length 39 .…”

Section: Discussionmentioning

confidence: 99%

“…Influential day-to-day factors may alter an individual's ability to complete certain assessments ultimately skewing their functional score and disease severity picture. Õunpuu et al 39 F I G U R E 5 An analysis of fatigue by comparing stride duration during the first and last quarter of 6MWT for Charcot-Marie-Tooth disease type 1A participants (top) and healthy controls (bottom). The x-axis shows the participant ID and y-axis shows stride duration in seconds.…”

Section: Discussionmentioning

confidence: 99%

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Disease‐specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot–Marie–Tooth disease type 1A

Dinesh

White

Baker

et al. 2023

J Peripheral Nervous Sys

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Background/AimsCharcot–Marie–Tooth Disease type 1A (CMT1A), the most common inherited peripheral neuropathy, is characterized by progressive sensory loss and weakness, which results in impaired mobility. Increased understanding of the genetics and pathophysiology of CMT1A has led to development of potential therapeutic agents, necessitating clinical trial readiness. Wearable sensors may provide useful outcome measures for future trials.MethodsIndividuals with CMT1A and unaffected controls were recruited for this 12‐month study. Participants wore sensors for in‐clinic assessments and at‐home, from which activity, gait, and balance metrics were derived. Mann–Whitney U tests were used to analyze group differences for activity, gait, and balance parameters. Test–retest reliability of gait and balance parameters and correlations of these parameters with clinical outcome assessments (COAs) were examined.ResultsThirty individuals, 15 CMT1A, and 15 controls, participated. Gait and balance metrics demonstrated moderate to excellent reliability. CMT1A participants had longer step durations (p < .001), shorter step lengths (p = .03), slower gait speeds (p < .001), and greater postural sway (p < .001) than healthy controls. Moderate correlations were found between CMT‐Functional Outcome Measure and step length (r = −0.59; p = .02), and gait speed (r = 0.64; p = .01); 11 out of 15 CMT1A participants demonstrated significant increases in stride duration between the first and last quarter of the 6‐min walk test, suggesting fatigue.InterpretationIn this initial study, gait and balance metrics derived from wearable sensors were reliable and associated with COAs in individuals with CMT1A. Larger longitudinal studies are needed to confirm our findings and evaluate sensitivity and utility of these disease‐specific algorithms for clinical trial use.

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The impact of orthoses on gait in children with Charcot-Marie-Tooth disease

Cited by 7 publications

References 18 publications

Effect of Ankle-Foot Orthoses in Pediatric Patients with Hereditary Motor-Sensory Neuropathy: A Case Series Study

Effect of Ankle-Foot Orthoses in Pediatric Patients with Hereditary Motor-Sensory Neuropathy: A Case Series Study

Clinical practice guideline for the management of paediatric Charcot-Marie-Tooth disease

Disease‐specific wearable sensor algorithms for profiling activity, gait, and balance in individuals with Charcot–Marie–Tooth disease type 1A

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