Epilepsy in LAMA2-related muscular dystrophy: An electro-clinico-radiological characterization.


Journal

Epilepsia
ISSN: 1528-1167
Titre abrégé: Epilepsia
Pays: United States
ID NLM: 2983306R

Informations de publication

Date de publication:
05 2020
Historique:
received: 14 11 2019
revised: 02 03 2020
accepted: 09 03 2020
pubmed: 9 4 2020
medline: 1 12 2020
entrez: 9 4 2020
Statut: ppublish

Résumé

To delineate the epileptic phenotype of LAMA2-related muscular dystrophy (MD) and correlate it with the neuroradiological and muscle biopsy findings, as well as the functional motor phenotype. Clinical, electrophysiological, neuroradiological, and histopathological data of 25 patients with diagnosis of LAMA2-related MD were analyzed. Epilepsy occurred in 36% of patients with LAMA2-related MD. Mean age at first seizure was 8 years. The most common presenting seizure type was focal-onset seizures with or without impaired awareness. Visual aura and autonomic signs, including vomiting, were frequently reported. Despite a certain degree of variability, bilateral occipital or temporo-occipital epileptiform abnormalities were by far the most commonly observed. Refractory epilepsy was found in 75% of these patients. Epilepsy in LAMA2-related MD was significantly more prevalent in those patients in whom the cortical malformations were more extensive. In contrast, the occurrence of epilepsy was not found to be associated with the patients' motor ability, the size of their white matter abnormalities, or the amount of residual merosin expressed on muscle. The epileptic phenotype of LAMA2-related MD is characterized by focal seizures with prominent visual and autonomic features associated with EEG abnormalities that predominate in the posterior quadrants. A consistent correlation between epileptic phenotype and neuroimaging was identified, suggesting that the extension of the polymicrogyria may serve as a predictor of epilepsy occurrence.

Identifiants

pubmed: 32266982
doi: 10.1111/epi.16493
doi:

Substances chimiques

Anticonvulsants 0

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Pagination

971-983

Subventions

Organisme : Instituto de Salud Carlos III, Río Hortega
ID : CM17/00054
Pays : International
Organisme : Instituto de Salud Carlos III, Río Hortega
ID : PI15/01898
Pays : International
Organisme : Fundación Mutua Madrileña
Pays : International
Organisme : European Regional Development Fund (FEDER a way to achieve Europe)
Pays : International

Informations de copyright

© 2020 International League Against Epilepsy.

Références

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Auteurs

Daniel Natera-de Benito (D)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Jordi Muchart (J)

Department of Radiology, Hospital Sant Joan de Déu, Barcelona, Spain.

Debora Itzep (D)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Carlos Ortez (C)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Lidia González-Quereda (L)

Department of Genetics, Hospital de la Santa Creu i Sant Pau and CIBERER U705, Barcelona, Spain.

Pía Gallano (P)

Department of Genetics, Hospital de la Santa Creu i Sant Pau and CIBERER U705, Barcelona, Spain.

Alia Ramirez (A)

Unit of Epilepsy, Sleep and Neurophysiology, Neuropaediatrics Department, Hospital Sant Joan de Déu, Barcelona, Spain.

Javier Aparicio (J)

Unit of Epilepsy, Sleep and Neurophysiology, Neuropaediatrics Department, Hospital Sant Joan de Déu, Barcelona, Spain.

Jana Domínguez-Carral (J)

Unit of Epilepsy, Sleep and Neurophysiology, Neuropaediatrics Department, Hospital Sant Joan de Déu, Barcelona, Spain.

Laura Carrera-García (L)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Jessica Expósito-Escudero (J)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Nathalia Pardo Cardozo (N)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Daniel Cuadras (D)

Statistics Department, Fundació Sant Joan de Déu, Barcelona, Spain.

Anna Codina (A)

Department of Pathology, Hospital Sant Joan de Déu, Barcelona, Spain.

Cristina Jou (C)

Department of Pathology, Hospital Sant Joan de Déu, Barcelona, Spain.

Cecilia Jimenez-Mallebrera (C)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Francesc Palau (F)

Department of Genetic and Molecular Medicine, Hospital Sant Joan de Déu, Barcelona, Spain.
Laboratory of Neurogenetics and Molecular Medicine, Institut de Recerca Sant Joan de Déu, Barcelona, Spain.
Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Barcelona, Spain.
Institute of Medicine and Dermatology, Hospital Clínic and Division of Pediatrics, University of Barcelona School of Medicine and Health Sciences, Barcelona, Spain.

Jaume Colomer (J)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Alexis Arzimanoglou (A)

Unit of Epilepsy, Sleep and Neurophysiology, Neuropaediatrics Department, Hospital Sant Joan de Déu, Barcelona, Spain.
Epileptology, Sleep Disorders and Functional Pediatric Neurology, Member of ERN-EpiCARE; HFME, Hospices Civils de Lyon, Bron, France.

Andrés Nascimento (A)

Neuromuscular Unit, Neuropaediatrics Department, Institut de Recerca Hospital Sant Joan de Déu and CIBERER U703, Barcelona, Spain.

Victoria San Antonio-Arce (V)

Unit of Epilepsy, Sleep and Neurophysiology, Neuropaediatrics Department, Hospital Sant Joan de Déu, Barcelona, Spain.

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