Is overwork weakness relevant in Charcot-Marie-Tooth disease?

G. Piscosquito, Mary M. Reilly, A. Schenone, G. M. Fabrizi, Tiziana Cavallaro, L. Santoro, G. Vita, A. Quattrone, L. Padua, F. Gemignani, F. Visioli, M. Laurà, D. Calabrese, R. A C Hughes, D. Radice, A. Solari, D. Pareyson, CMT-TRIAAL & CMT-TRAUK Group

Research output: Contribution to journalArticle

Abstract

BACKGROUND: In overwork weakness (OW), muscles are increasingly weakened by exercise, work or daily activities. Although it is a well-established phenomenon in several neuromuscular disorders, it is debated whether it occurs in Charcot-Marie-Tooth disease (CMT). Dominant limb muscles undergo a heavier overload than non-dominant and therefore if OW occurs we would expect them to become weaker. Four previous studies, comparing dominant and non-dominant hand strength in CMT series employing manual testing or myometry, gave contradictory results. Moreover, none of them examined the behaviour of lower limb muscles. METHODS: We tested the OW hypothesis in 271 CMT1A adult patients by comparing bilateral intrinsic hand and leg muscle strength with manual testing as well as manual dexterity. RESULTS: We found no significant difference between sides for the strength of first dorsal interosseous, abductor pollicis brevis, anterior tibialis and triceps surae. Dominant side muscles did not become weaker than non-dominant with increasing age and disease severity (assessed with the CMT Neuropathy Score); in fact, the dominant triceps surae was slightly stronger than the non-dominant with increasing age and disease severity. DISCUSSION: Our data does not support the OW hypothesis and the consequent harmful effect of exercise in patients with CMT1A. Physical activity should be encouraged, and rehabilitation remains the most effective treatment for CMT patients. Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://group.bmj.com/group/rights-licensing/permissions.
Original languageEnglish
Pages (from-to)1354-1358
Number of pages5
JournalJournal of Neurology, Neurosurgery and Psychiatry
Volume85
Issue number12
DOIs
Publication statusPublished - Mar 2014

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Charcot-Marie-Tooth Disease
Exercise
Licensure
Muscles
Muscle Weakness
Muscle Strength
Hand Strength
Lower Extremity
Leg
Rehabilitation
Extremities
Hand

Keywords

  • CLINICAL NEUROLOGY
  • HMSN (CHARCOT-MARIE-TOOTH)
  • NEUROGENETICS
  • NEUROPATHY
  • REHABILITATION
  • adolescent
  • adult
  • aged
  • complication
  • Cumulative Trauma Disorders
  • female
  • hand strength
  • hemispheric dominance
  • hereditary motor sensory neuropathy
  • human
  • male
  • middle aged
  • muscle strength
  • muscle weakness
  • pathophysiology
  • physiology
  • skeletal muscle
  • young adult
  • Adolescent
  • Adult
  • Aged
  • Charcot-Marie-Tooth Disease
  • Female
  • Functional Laterality
  • Hand Strength
  • Humans
  • Male
  • Middle Aged
  • Muscle Strength
  • Muscle Weakness
  • Muscle, Skeletal
  • Young Adult

Cite this

Piscosquito, G., Reilly, M. M., Schenone, A., Fabrizi, G. M., Cavallaro, T., Santoro, L., ... Group, CMT-TRIAAL. . CMT-TRAUK. (2014). Is overwork weakness relevant in Charcot-Marie-Tooth disease? Journal of Neurology, Neurosurgery and Psychiatry, 85(12), 1354-1358. https://doi.org/10.1136/jnnp-2014-307598

Is overwork weakness relevant in Charcot-Marie-Tooth disease? / Piscosquito, G.; Reilly, Mary M.; Schenone, A.; Fabrizi, G. M.; Cavallaro, Tiziana; Santoro, L.; Vita, G.; Quattrone, A.; Padua, L.; Gemignani, F.; Visioli, F.; Laurà, M.; Calabrese, D.; Hughes, R. A C; Radice, D.; Solari, A.; Pareyson, D.; Group, CMT-TRIAAL & CMT-TRAUK.

In: Journal of Neurology, Neurosurgery and Psychiatry, Vol. 85, No. 12, 03.2014, p. 1354-1358.

Research output: Contribution to journalArticle

Piscosquito, G, Reilly, MM, Schenone, A, Fabrizi, GM, Cavallaro, T, Santoro, L, Vita, G, Quattrone, A, Padua, L, Gemignani, F, Visioli, F, Laurà, M, Calabrese, D, Hughes, RAC, Radice, D, Solari, A, Pareyson, D & Group, CMT-TRIAALCMT-TRAUK 2014, 'Is overwork weakness relevant in Charcot-Marie-Tooth disease?', Journal of Neurology, Neurosurgery and Psychiatry, vol. 85, no. 12, pp. 1354-1358. https://doi.org/10.1136/jnnp-2014-307598
Piscosquito, G. ; Reilly, Mary M. ; Schenone, A. ; Fabrizi, G. M. ; Cavallaro, Tiziana ; Santoro, L. ; Vita, G. ; Quattrone, A. ; Padua, L. ; Gemignani, F. ; Visioli, F. ; Laurà, M. ; Calabrese, D. ; Hughes, R. A C ; Radice, D. ; Solari, A. ; Pareyson, D. ; Group, CMT-TRIAAL & CMT-TRAUK. / Is overwork weakness relevant in Charcot-Marie-Tooth disease?. In: Journal of Neurology, Neurosurgery and Psychiatry. 2014 ; Vol. 85, No. 12. pp. 1354-1358.
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abstract = "BACKGROUND: In overwork weakness (OW), muscles are increasingly weakened by exercise, work or daily activities. Although it is a well-established phenomenon in several neuromuscular disorders, it is debated whether it occurs in Charcot-Marie-Tooth disease (CMT). Dominant limb muscles undergo a heavier overload than non-dominant and therefore if OW occurs we would expect them to become weaker. Four previous studies, comparing dominant and non-dominant hand strength in CMT series employing manual testing or myometry, gave contradictory results. Moreover, none of them examined the behaviour of lower limb muscles. METHODS: We tested the OW hypothesis in 271 CMT1A adult patients by comparing bilateral intrinsic hand and leg muscle strength with manual testing as well as manual dexterity. RESULTS: We found no significant difference between sides for the strength of first dorsal interosseous, abductor pollicis brevis, anterior tibialis and triceps surae. Dominant side muscles did not become weaker than non-dominant with increasing age and disease severity (assessed with the CMT Neuropathy Score); in fact, the dominant triceps surae was slightly stronger than the non-dominant with increasing age and disease severity. DISCUSSION: Our data does not support the OW hypothesis and the consequent harmful effect of exercise in patients with CMT1A. Physical activity should be encouraged, and rehabilitation remains the most effective treatment for CMT patients. Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://group.bmj.com/group/rights-licensing/permissions.",
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TY - JOUR

T1 - Is overwork weakness relevant in Charcot-Marie-Tooth disease?

AU - Piscosquito, G.

AU - Reilly, Mary M.

AU - Schenone, A.

AU - Fabrizi, G. M.

AU - Cavallaro, Tiziana

AU - Santoro, L.

AU - Vita, G.

AU - Quattrone, A.

AU - Padua, L.

AU - Gemignani, F.

AU - Visioli, F.

AU - Laurà, M.

AU - Calabrese, D.

AU - Hughes, R. A C

AU - Radice, D.

AU - Solari, A.

AU - Pareyson, D.

AU - Group, CMT-TRIAAL & CMT-TRAUK

N1 - Cited By :4 Export Date: 11 May 2017 Funding details: G0802497, MRC, Medical Research Council Funding details: MR/K000608/1, MRC, Medical Research Council

PY - 2014/3

Y1 - 2014/3

N2 - BACKGROUND: In overwork weakness (OW), muscles are increasingly weakened by exercise, work or daily activities. Although it is a well-established phenomenon in several neuromuscular disorders, it is debated whether it occurs in Charcot-Marie-Tooth disease (CMT). Dominant limb muscles undergo a heavier overload than non-dominant and therefore if OW occurs we would expect them to become weaker. Four previous studies, comparing dominant and non-dominant hand strength in CMT series employing manual testing or myometry, gave contradictory results. Moreover, none of them examined the behaviour of lower limb muscles. METHODS: We tested the OW hypothesis in 271 CMT1A adult patients by comparing bilateral intrinsic hand and leg muscle strength with manual testing as well as manual dexterity. RESULTS: We found no significant difference between sides for the strength of first dorsal interosseous, abductor pollicis brevis, anterior tibialis and triceps surae. Dominant side muscles did not become weaker than non-dominant with increasing age and disease severity (assessed with the CMT Neuropathy Score); in fact, the dominant triceps surae was slightly stronger than the non-dominant with increasing age and disease severity. DISCUSSION: Our data does not support the OW hypothesis and the consequent harmful effect of exercise in patients with CMT1A. Physical activity should be encouraged, and rehabilitation remains the most effective treatment for CMT patients. Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://group.bmj.com/group/rights-licensing/permissions.

AB - BACKGROUND: In overwork weakness (OW), muscles are increasingly weakened by exercise, work or daily activities. Although it is a well-established phenomenon in several neuromuscular disorders, it is debated whether it occurs in Charcot-Marie-Tooth disease (CMT). Dominant limb muscles undergo a heavier overload than non-dominant and therefore if OW occurs we would expect them to become weaker. Four previous studies, comparing dominant and non-dominant hand strength in CMT series employing manual testing or myometry, gave contradictory results. Moreover, none of them examined the behaviour of lower limb muscles. METHODS: We tested the OW hypothesis in 271 CMT1A adult patients by comparing bilateral intrinsic hand and leg muscle strength with manual testing as well as manual dexterity. RESULTS: We found no significant difference between sides for the strength of first dorsal interosseous, abductor pollicis brevis, anterior tibialis and triceps surae. Dominant side muscles did not become weaker than non-dominant with increasing age and disease severity (assessed with the CMT Neuropathy Score); in fact, the dominant triceps surae was slightly stronger than the non-dominant with increasing age and disease severity. DISCUSSION: Our data does not support the OW hypothesis and the consequent harmful effect of exercise in patients with CMT1A. Physical activity should be encouraged, and rehabilitation remains the most effective treatment for CMT patients. Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://group.bmj.com/group/rights-licensing/permissions.

KW - CLINICAL NEUROLOGY

KW - HMSN (CHARCOT-MARIE-TOOTH)

KW - NEUROGENETICS

KW - NEUROPATHY

KW - REHABILITATION

KW - adolescent

KW - adult

KW - aged

KW - complication

KW - Cumulative Trauma Disorders

KW - female

KW - hand strength

KW - hemispheric dominance

KW - hereditary motor sensory neuropathy

KW - human

KW - male

KW - middle aged

KW - muscle strength

KW - muscle weakness

KW - pathophysiology

KW - physiology

KW - skeletal muscle

KW - young adult

KW - Adolescent

KW - Adult

KW - Aged

KW - Charcot-Marie-Tooth Disease

KW - Female

KW - Functional Laterality

KW - Hand Strength

KW - Humans

KW - Male

KW - Middle Aged

KW - Muscle Strength

KW - Muscle Weakness

KW - Muscle, Skeletal

KW - Young Adult

U2 - 10.1136/jnnp-2014-307598

DO - 10.1136/jnnp-2014-307598

M3 - Article

VL - 85

SP - 1354

EP - 1358

JO - Journal of Neurology, Neurosurgery and Psychiatry

JF - Journal of Neurology, Neurosurgery and Psychiatry

SN - 0022-3050

IS - 12

ER -