Association of Body Mass Index With Disease Progression in Children With Charcot-Marie-Tooth Disease.

Adult Humans Child Body Mass Index Overweight / complications Thinness / epidemiology Charcot-Marie-Tooth Disease / complications Obesity / complications Disease Progression

Journal

Neurology
ISSN: 1526-632X
Titre abrégé: Neurology
Pays: United States
ID NLM: 0401060

Informations de publication

Date de publication:
15 08 2023
Historique:
received: 14 09 2022
accepted: 20 04 2023
pmc-release: 15 08 2024
medline: 16 8 2023
pubmed: 29 6 2023
entrez: 28 6 2023
Statut: ppublish

Résumé

The aim of this study was to evaluate the impact of body mass index (BMI) on disease progression over 2 years in children with Charcot-Marie-Tooth disease (CMT). BMI was classified in 242 participants aged 3-20 years with CMT enrolled in the Inherited Neuropathy Consortium, using the International Obesity Task Force (based on adult BMI values, kg/m At baseline, compared with individuals being of a healthy weight (mean CMTPedS 15.48, SD 9.22), children who were severely underweight (mean CMTPedS difference 9.03, 95% CI 0.94-17.12; Children with CMT who were severely underweight, underweight, or obese exhibited greater disability at baseline. Over the 2-year period in those whose BMI remained stable, severely underweight children deteriorated at the fastest rate. For children who changed BMI categories over the 2 years, CMTPedS scores deteriorated faster in children who became overweight/obese. Interventions that maintain or improve BMI toward healthy weight may reduce disability in children with CMT.

Sections du résumé

BACKGROUND AND OBJECTIVES
The aim of this study was to evaluate the impact of body mass index (BMI) on disease progression over 2 years in children with Charcot-Marie-Tooth disease (CMT).
METHODS
BMI was classified in 242 participants aged 3-20 years with CMT enrolled in the Inherited Neuropathy Consortium, using the International Obesity Task Force (based on adult BMI values, kg/m
RESULTS
At baseline, compared with individuals being of a healthy weight (mean CMTPedS 15.48, SD 9.22), children who were severely underweight (mean CMTPedS difference 9.03, 95% CI 0.94-17.12;
DISCUSSION
Children with CMT who were severely underweight, underweight, or obese exhibited greater disability at baseline. Over the 2-year period in those whose BMI remained stable, severely underweight children deteriorated at the fastest rate. For children who changed BMI categories over the 2 years, CMTPedS scores deteriorated faster in children who became overweight/obese. Interventions that maintain or improve BMI toward healthy weight may reduce disability in children with CMT.

Identifiants

DOI: 10.1212/WNL.0000000000207488 PMID: 37380432 PMC: PMC10437011
pubmed: 37380432
pii: WNL.0000000000207488
doi: 10.1212/WNL.0000000000207488
pmc: PMC10437011
doi:

Types de publication

Journal Article Research Support, N.I.H., Extramural

Langues

eng

Sous-ensembles de citation

IM

Pagination

e717-e727

Subventions

Organisme : NINDS NIH HHS
ID : U54 NS065712
Pays : United States

Informations de copyright

© 2023 American Academy of Neurology.

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Auteurs

Gabrielle A Donlevy (GA)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY. gabrielle.donlevy@sydney.edu.au.
ORCID: 0000-0002-4708-5879

Kayla M D Cornett (KMD)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Sarah P Garnett (SP)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0001-7552-0789

Rosemary Shy (R)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Timothy Estilow (T)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Sabrina W Yum (SW)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0002-5243-5546

Kimberly Anderson (K)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Davide Pareyson (D)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0001-6854-765X

Isabella Moroni (I)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Francesco Muntoni (F)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0002-9102-5232

Mary M Reilly (MM)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Richard S Finkel (RS)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0002-9351-7054

David N Herrmann (DN)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0002-9023-7970

Katy J Eichinger (KJ)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.

Michael E Shy (ME)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0001-8460-6971

Joshua Burns (J)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0001-7936-623X

Manoj P Menezes (MP)

From the Faculty of Medicine and Health & Children's Hospital at Westmead (G.A.D., K.M.D.C., S.P.G., J.B., M.P.M.), University of Sydney, Australia; Department of Neurology (R.S., M.E.S.), and Department of Pediatrics (T.E., S.W.Y.), Carver College of Medicine, University of Iowa, Iowa City; Division of Neurology (T.E., S.W.Y.), The Children's Hospital of Philadelphia, PA; Department of Neurology (T.E., S.W.Y.), Perelman School of Medicine, University of Pennsylvania, Philadelphia; Department of Occupational Therapy (K.A.), Children's Hospital of Philadelphia, PA; Fondazione IRCCS (D.P., I.M.), Istituto Neurologico Carlo Besta, Milan, Italy; University College London Institute of Child Health & Great Ormond Street Hospital (F.M.); Department of Neuromuscular Diseases (M.M.R.), University College London Institute of Neurology, Queen Square, United Kingdom; Center for Experimental Neurotherapeutics (Pediatrics) (R.S.F.), St. Jude Children's Research Hospital, Memphis, TN; and Department of Neurology (D.N.H., K.J.E.), University of Rochester, NY.
ORCID: 0000-0003-3160-6112

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Classifications MeSH

questionsmedicales.fr › Personnes › Tranches d'âge › Adulte › Association of Body Mass Index With Disease...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › Association of Body Mass Index With Disease...
questionsmedicales.fr › Personnes › Tranches d'âge › Enfant › Association of Body Mass Index With Disease...
questionsmedicales.fr › Environnement et santé publique › Santé publique › Mesures épidémiologiques › Biométrie › Anthropométrie › Indice de masse corporelle › Association of Body Mass Index With Disease...
questionsmedicales.fr › Phénomènes physiologiques › Constitution physique › Apparence corporelle › Surpoids › Association of Body Mass Index With Disease...
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