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

G. Piscosquito, M. M. Reilly, A. Schenone, G. M. Fabrizi, T. 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, C. Besta, C. Marchesi, E. Salsano & 40 others L. Nanetti, C. Marelli, V. Scaioli, C. Ciano, M. Rimoldi, G. Lauria, E. Rizzetto, F. Camozzi, E. Narciso, M. Grandis, M. Monti-Bragadin, L. Nobbio, A. Casano, L. Bertolasi, I. Cabrini, K. Corrà, N. Rizzuto, F. Manganelli, C. Pisciotta, M. Nolano, A. Mazzeo, R. Di Leo, G. Majorana, M. Russo, P. Valentino, R. Nisticò, D. Pirritano, A. Lucisano, M. Canino, C. Pazzaglia, G. Granata, M. Foschini, F. Brindani, F. Vitetta, I. Allegri, P. Bogani, J. Blake, M. Koltzenburg, E. Hutton, M. Lunn

Research output: Contribution to journalArticle

15 Citations (Scopus)

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.

Original languageEnglish
JournalJournal of Neurology, Neurosurgery and Psychiatry
DOIs
Publication statusAccepted/In press - Mar 21 2014

Fingerprint

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

ASJC Scopus subject areas

  • Arts and Humanities (miscellaneous)
  • Surgery
  • Psychiatry and Mental health
  • Clinical Neurology

Cite this

Piscosquito, G., Reilly, M. M., Schenone, A., Fabrizi, G. M., Cavallaro, T., Santoro, L., ... Lunn, M. (Accepted/In press). Is overwork weakness relevant in Charcot-Marie-Tooth disease? Journal of Neurology, Neurosurgery and Psychiatry. https://doi.org/10.1136/jnnp-2014-307598

Is overwork weakness relevant in Charcot-Marie-Tooth disease? / Piscosquito, G.; Reilly, M. M.; Schenone, A.; Fabrizi, G. M.; Cavallaro, T.; 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.; Besta, C.; Marchesi, C.; Salsano, E.; Nanetti, L.; Marelli, C.; Scaioli, V.; Ciano, C.; Rimoldi, M.; Lauria, G.; Rizzetto, E.; Camozzi, F.; Narciso, E.; Grandis, M.; Monti-Bragadin, M.; Nobbio, L.; Casano, A.; Bertolasi, L.; Cabrini, I.; Corrà, K.; Rizzuto, N.; Manganelli, F.; Pisciotta, C.; Nolano, M.; Mazzeo, A.; Di Leo, R.; Majorana, G.; Russo, M.; Valentino, P.; Nisticò, R.; Pirritano, D.; Lucisano, A.; Canino, M.; Pazzaglia, C.; Granata, G.; Foschini, M.; Brindani, F.; Vitetta, F.; Allegri, I.; Bogani, P.; Blake, J.; Koltzenburg, M.; Hutton, E.; Lunn, M.

In: Journal of Neurology, Neurosurgery and Psychiatry, 21.03.2014.

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, Besta, C, Marchesi, C, Salsano, E, Nanetti, L, Marelli, C, Scaioli, V, Ciano, C, Rimoldi, M, Lauria, G, Rizzetto, E, Camozzi, F, Narciso, E, Grandis, M, Monti-Bragadin, M, Nobbio, L, Casano, A, Bertolasi, L, Cabrini, I, Corrà, K, Rizzuto, N, Manganelli, F, Pisciotta, C, Nolano, M, Mazzeo, A, Di Leo, R, Majorana, G, Russo, M, Valentino, P, Nisticò, R, Pirritano, D, Lucisano, A, Canino, M, Pazzaglia, C, Granata, G, Foschini, M, Brindani, F, Vitetta, F, Allegri, I, Bogani, P, Blake, J, Koltzenburg, M, Hutton, E & Lunn, M 2014, 'Is overwork weakness relevant in Charcot-Marie-Tooth disease?', Journal of Neurology, Neurosurgery and Psychiatry. https://doi.org/10.1136/jnnp-2014-307598
Piscosquito, G. ; Reilly, M. M. ; Schenone, A. ; Fabrizi, G. M. ; Cavallaro, T. ; 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. ; Besta, C. ; Marchesi, C. ; Salsano, E. ; Nanetti, L. ; Marelli, C. ; Scaioli, V. ; Ciano, C. ; Rimoldi, M. ; Lauria, G. ; Rizzetto, E. ; Camozzi, F. ; Narciso, E. ; Grandis, M. ; Monti-Bragadin, M. ; Nobbio, L. ; Casano, A. ; Bertolasi, L. ; Cabrini, I. ; Corrà, K. ; Rizzuto, N. ; Manganelli, F. ; Pisciotta, C. ; Nolano, M. ; Mazzeo, A. ; Di Leo, R. ; Majorana, G. ; Russo, M. ; Valentino, P. ; Nisticò, R. ; Pirritano, D. ; Lucisano, A. ; Canino, M. ; Pazzaglia, C. ; Granata, G. ; Foschini, M. ; Brindani, F. ; Vitetta, F. ; Allegri, I. ; Bogani, P. ; Blake, J. ; Koltzenburg, M. ; Hutton, E. ; Lunn, M. / Is overwork weakness relevant in Charcot-Marie-Tooth disease?. In: Journal of Neurology, Neurosurgery and Psychiatry. 2014.
@article{644667f3267849a391c9e662b04231df,
title = "Is overwork weakness relevant in Charcot-Marie-Tooth disease?",
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.",
author = "G. Piscosquito and Reilly, {M. M.} and A. Schenone and Fabrizi, {G. M.} and T. Cavallaro and L. Santoro and G. Vita and A. Quattrone and L. Padua and F. Gemignani and F. Visioli and M. Laur{\`a} and D. Calabrese and Hughes, {R. A C} and D. Radice and A. Solari and D. Pareyson and C. Besta and C. Marchesi and E. Salsano and L. Nanetti and C. Marelli and V. Scaioli and C. Ciano and M. Rimoldi and G. Lauria and E. Rizzetto and F. Camozzi and E. Narciso and M. Grandis and M. Monti-Bragadin and L. Nobbio and A. Casano and L. Bertolasi and I. Cabrini and K. Corr{\`a} and N. Rizzuto and F. Manganelli and C. Pisciotta and M. Nolano and A. Mazzeo and {Di Leo}, R. and G. Majorana and M. Russo and P. Valentino and R. Nistic{\`o} and D. Pirritano and A. Lucisano and M. Canino and C. Pazzaglia and G. Granata and M. Foschini and F. Brindani and F. Vitetta and I. Allegri and P. Bogani and J. Blake and M. Koltzenburg and E. Hutton and M. Lunn",
year = "2014",
month = "3",
day = "21",
doi = "10.1136/jnnp-2014-307598",
language = "English",
journal = "Journal of Neurology, Neurosurgery and Psychiatry",
issn = "0022-3050",
publisher = "BMJ Publishing Group",

}

TY - JOUR

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

AU - Piscosquito, G.

AU - Reilly, M. M.

AU - Schenone, A.

AU - Fabrizi, G. M.

AU - Cavallaro, T.

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 - Besta, C.

AU - Marchesi, C.

AU - Salsano, E.

AU - Nanetti, L.

AU - Marelli, C.

AU - Scaioli, V.

AU - Ciano, C.

AU - Rimoldi, M.

AU - Lauria, G.

AU - Rizzetto, E.

AU - Camozzi, F.

AU - Narciso, E.

AU - Grandis, M.

AU - Monti-Bragadin, M.

AU - Nobbio, L.

AU - Casano, A.

AU - Bertolasi, L.

AU - Cabrini, I.

AU - Corrà, K.

AU - Rizzuto, N.

AU - Manganelli, F.

AU - Pisciotta, C.

AU - Nolano, M.

AU - Mazzeo, A.

AU - Di Leo, R.

AU - Majorana, G.

AU - Russo, M.

AU - Valentino, P.

AU - Nisticò, R.

AU - Pirritano, D.

AU - Lucisano, A.

AU - Canino, M.

AU - Pazzaglia, C.

AU - Granata, G.

AU - Foschini, M.

AU - Brindani, F.

AU - Vitetta, F.

AU - Allegri, I.

AU - Bogani, P.

AU - Blake, J.

AU - Koltzenburg, M.

AU - Hutton, E.

AU - Lunn, M.

PY - 2014/3/21

Y1 - 2014/3/21

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.

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.

UR - http://www.scopus.com/inward/record.url?scp=84896411035&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84896411035&partnerID=8YFLogxK

U2 - 10.1136/jnnp-2014-307598

DO - 10.1136/jnnp-2014-307598

M3 - Article

JO - Journal of Neurology, Neurosurgery and Psychiatry

JF - Journal of Neurology, Neurosurgery and Psychiatry

SN - 0022-3050

ER -