Muscle MRI in a large cohort of patients with oculopharyngeal muscular dystrophy.


Journal

Journal of neurology, neurosurgery, and psychiatry
ISSN: 1468-330X
Titre abrégé: J Neurol Neurosurg Psychiatry
Pays: England
ID NLM: 2985191R

Informations de publication

Date de publication:
05 2019
Historique:
received: 04 09 2018
revised: 13 11 2018
accepted: 19 11 2018
pubmed: 12 12 2018
medline: 20 3 2020
entrez: 12 12 2018
Statut: ppublish

Résumé

Oculopharyngeal muscular dystrophy (OPMD) is a genetic disorder caused by an abnormal expansion of GCN triplets within the We present cross-sectional, T1-weighted muscle MRI and CT-scan data from 168 patients with genetically confirmed OPMD. We have analysed the pattern of muscle involvement in the disease using hierarchical analysis and presented it as heatmaps. Results of the scans were correlated with genetic and clinical data. Fatty replacement was identified in 96.7% of all symptomatic patients. The tongue, the We have described a pattern that can be considered characteristic of OPMD. An early combination of fat replacement in the tongue,

Sections du résumé

BACKGROUND AND OBJECTIVE
Oculopharyngeal muscular dystrophy (OPMD) is a genetic disorder caused by an abnormal expansion of GCN triplets within the
METHODS
We present cross-sectional, T1-weighted muscle MRI and CT-scan data from 168 patients with genetically confirmed OPMD. We have analysed the pattern of muscle involvement in the disease using hierarchical analysis and presented it as heatmaps. Results of the scans were correlated with genetic and clinical data.
RESULTS
Fatty replacement was identified in 96.7% of all symptomatic patients. The tongue, the
CONCLUSIONS
We have described a pattern that can be considered characteristic of OPMD. An early combination of fat replacement in the tongue,

Identifiants

pubmed: 30530568
pii: jnnp-2018-319578
doi: 10.1136/jnnp-2018-319578
doi:

Types de publication

Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

576-585

Informations de copyright

© Author(s) (or their employer(s)) 2019. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

Auteurs

Alicia Alonso-Jimenez (A)

Neuromuscular Disorders Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universidad Autónoma de Barcelona, Barcelona, Spain.

Rosemarie H M J M Kroon (RHMJM)

Rehabilitation Department, Radboud University Medical Center, Nijmegen, The Netherlands.

Aida Alejaldre-Monforte (A)

Neurology Department, Hospital de Sant Joan Despí Moisès Broggi, Barcelona, Spain.

Claudia Nuñez-Peralta (C)

Radiology Department, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain.

Corinne G C Horlings (CGC)

Neurology Department, Radboud university Medical Center, Nijmegen, The Netherlands.

Baziel G M van Engelen (BGM)

Neurology Department, Radboud university Medical Center, Nijmegen, The Netherlands.

Montse Olivé (M)

Pathology Department (Neuropathology), Neuromuscular Disorders Unit, IDIBELL, Hospital de Bellvitge, Barcelona, Spain.

Laura González (L)

Pathology Department (Neuropathology), Neuromuscular Disorders Unit, IDIBELL, Hospital de Bellvitge, Barcelona, Spain.

Enric Verges-Gil (E)

Pathology Department (Neuropathology), Neuromuscular Disorders Unit, IDIBELL, Hospital de Bellvitge, Barcelona, Spain.

Carmen Paradas (C)

Neuromuscular Disorders Unit, Neurology Department, Hospital Universitario Virgen del Rocío, Instituto de Biomedicina de Sevilla, Sevilla, Spain.

Celedonio Márquez (C)

Neuromuscular Disorders Unit, Neurology Department, Hospital Universitario Virgen del Rocío, Instituto de Biomedicina de Sevilla, Sevilla, Spain.

Matteo Garibaldi (M)

Neuromuscular Disorders Unit, Department of Neurology, Mental Health and Sensory Organs (NESMOS), SAPIENZA University of Rome, Ospedale Sant'Andrea, Rome, Italy.

Pía Gallano (P)

Genetic Department, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

Maria José Rodriguez (MJ)

Genetic Department, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain.

Lidia Gonzalez-Quereda (L)

Genetic Department, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

Cristina Dominguez Gonzalez (C)

Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.
Neuromuscular Disorders Unit, Neurology Department, Hospital Universitario 12 de Octubre, Madrid, Spain.

John Vissing (J)

Copenhagen Neuromuscular Center, Rigshospitalet, University of Copenhagen, Copenhagen, Denmark.

Freja Fornander (F)

Copenhagen Neuromuscular Center, Rigshospitalet, University of Copenhagen, Copenhagen, Denmark.

Anne-Sofie Vibæk Eisum (AV)

Copenhagen Neuromuscular Center, Rigshospitalet, University of Copenhagen, Copenhagen, Denmark.

Tania García-Sobrino (T)

Neurology Department, Hospital Clínico, Santiago de Compostela, Spain.

Julio Pardo (J)

Neurology Department, Hospital Clínico, Santiago de Compostela, Spain.

Roberto García-Figueiras (R)

Radiology Department, Hospital Clínico, Santiago de Compostela, Spain.

Nuria Muelas (N)

Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.
Neuromuscular Research Unit, Neurology Department, Instituto de Investigación Sanitaria la Fe, Hospital Universitari i Politécnic La Fe, Valencia, Spain.

Juan Jesús Vilchez (JJ)

Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.
Neuromuscular Research Unit, Neurology Department, Instituto de Investigación Sanitaria la Fe, Hospital Universitari i Politécnic La Fe, Valencia, Spain.

Solange Kapetanovic (S)

Neurology Department, Hospital de Basurto, Bilbao, Spain.

Giorgio Tasca (G)

Unità Operativa Complessa di Neurologia, Fondazione Policlinico Universitario A, Gemelli IRCCS, Roem, Italy.

Mauro Monforte (M)

Unità Operativa Complessa di Neurologia, Fondazione Policlinico Universitario A, Gemelli IRCCS, Roem, Italy.
Istituto di Neurologia, Università Cattolica del Sacro Cuore, Rome, Italy.

Enzo Ricci (E)

Unità Operativa Complessa di Neurologia, Fondazione Policlinico Universitario A, Gemelli IRCCS, Roem, Italy.
Istituto di Neurologia, Università Cattolica del Sacro Cuore, Rome, Italy.

María Teresa Gomez (MT)

Neurology Department, Hospital Universitario Reina Sofía, Córdoba, Spain.

Jorge Alfredo Bevilacqua (JA)

Departamento de Neurología y Neurocirugía, Hospital Clínico Universidad de Chile, Programa de Anatomía y Medicina Legal, Facultad de Medicina, Universidad de Chile, Santiago, Chile.

Jorge Diaz-Jara (J)

Centro de imagenología, Hospital Clínico Universidad de Chile, Universidad de Chile, Santiago, Chile.

Ivonne Ingrid Zamorano (II)

Servicio de Neurología, Hospital de Puerto Montt, Servicio de Salud del Reloncavi, Los Lagos Region, Chile.

Robert Yves Carlier (RY)

Assistance Publique des Hôpitaux de Paris (AP-HP), Service d'Imagerie Médicale, Pôle Neuro-locomoteur, Hôpital Raymond Poincaré, Garches, Hôpitaux Universitaires Paris-Ile-de-France Ouest, Garches, France.

Pascal Laforet (P)

Assistance Publique des Hôpitaux de Paris (AP-HP), Service de neurologie, Pôle Neuro-locomoteur, Hôpital Raymond Poincaré, Garches, Hôpitaux Universitaires Paris-Ile-de-France Ouest, Garches, France.

Ana Pelayo-Negro (A)

Neurology Department, University Hospital "Marqués de Valdecilla (IDIVAL)", University of Cantabria, and "Centro de Investigación Biomédica en Red de Enfermedades Neurodegenerativas (CIBERNED)", Santander, Spain.

Alba Ramos-Fransi (A)

Neurology Department, Hospital Germans Trias I Pujol, Barcelona, Spain.

Amaia Martínez (A)

Neurology Department, Hospital Galdakao, Vizkaia, Spain.

Chiara Marini-Bettolo (C)

The John Walton Muscular Dystrophy Research Centre, MRC Centre for Neuromuscular Diseases Institute of Genetic Medicine, University of Newcastle, Newcastle upon Tyne, UK.

Volker Straub (V)

The John Walton Muscular Dystrophy Research Centre, MRC Centre for Neuromuscular Diseases Institute of Genetic Medicine, University of Newcastle, Newcastle upon Tyne, UK.

Gerardo Gutiérrez (G)

Neurology Department, Hospital Infanta Sofía, San Sebastián de los Reyes, Spain.

Tanya Stojkovic (T)

Institute of Myology, Pitié-Salpêtrière Hospital, Paris, France.

María Asunción Martín (MA)

Neurology Department, Complejo asistencial hospitalario de Burgos, Burgos, Spain.

Germán Morís (G)

Neurology Department, Hospital Universitario Central de Asturias, Asturias, Spain.

Roberto Fernández-Torrón (R)

Neurology Department, Hospital Donostia, San Sebastián, Spain.
Neuromuscular Area, Neurology Service, Biodonostia Health Research Institute, Donostia University Hospital, Donostia-San Sebastián, Spain.

Adolfo Lopez De Munaín (A)

Neurology Department, Hospital Donostia, San Sebastián, Spain.
Neuromuscular Area, Neurology Service, Biodonostia Health Research Institute, Donostia University Hospital, Donostia-San Sebastián, Spain.

Elena Cortes-Vicente (E)

Neuromuscular Disorders Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universidad Autónoma de Barcelona, Barcelona, Spain.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

Luis Querol (L)

Neuromuscular Disorders Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universidad Autónoma de Barcelona, Barcelona, Spain.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

Ricardo Rojas-García (R)

Neuromuscular Disorders Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universidad Autónoma de Barcelona, Barcelona, Spain.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

Isabel Illa (I)

Neuromuscular Disorders Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universidad Autónoma de Barcelona, Barcelona, Spain.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

Jordi Diaz-Manera (J)

Neuromuscular Disorders Unit, Neurology Department, Hospital de la Santa Creu I Sant Pau, Universidad Autónoma de Barcelona, Barcelona, Spain jdiazm@santpau.cat.
Centro de Investigación Biomédica en Red en Enfermedades Raras (CIBERER), Valencia, Spain.

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