Early magnetic resonance imaging to detect presymptomatic leptomeningeal angioma in children with suspected Sturge-Weber syndrome.


Journal

Developmental medicine and child neurology
ISSN: 1469-8749
Titre abrégé: Dev Med Child Neurol
Pays: England
ID NLM: 0006761

Informations de publication

Date de publication:
02 2020
Historique:
accepted: 13 03 2019
pubmed: 3 5 2019
medline: 6 5 2020
entrez: 4 5 2019
Statut: ppublish

Résumé

We aimed to evaluate the contribution of early magnetic resonance imaging (MRI) for the presymptomatic diagnosis of Sturge-Weber syndrome (SWS) in infants with a facial port-wine birthmark (PWB). Asymptomatic infants with a facial PWB who performed a first MRI scan before 3 months and a second MRI scan after 9 months were included in this study. Leptomeningeal enhancement on T1-weighted imaging and four indirect signs of leptomeningeal angioma (choroid plexus enlargement, cerebral atrophy, signal inversion of the white matter with T2 hyposignal, and T1 hypersignal) were screened on the first MRI scan and correlated with clinical and/or radiological diagnosis of SWS. Thirteen of 30 included patients had SWS with leptomeningeal angioma. Eleven had a leptomeningeal enhancement on the first MRI scan and 10 had associated indirect signs. The presence of a direct or at least one indirect sign of leptomeningeal angioma on the first MRI scan confirmed the diagnosis of SWS with a sensitivity of 100 per cent (95% confidence interval 75-100%) and a specificity of 94 per cent (71-100%). Early diagnosis of SWS is possible on contrast-enhanced MRI performed in asymptomatic infants with a facial PWB before the age of 3 months. This early detection would help to select patients who may benefit from early neuroprotective intervention. Specific magnetic resonance imaging markers provide early diagnosis of leptomeningeal angioma in Sturge-Weber syndrome (SWS). Presymptomatic diagnosis of SWS should help to select patients for early therapy intervention.

Identifiants

pubmed: 31050360
doi: 10.1111/dmcn.14253
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

227-233

Informations de copyright

© 2019 Mac Keith Press.

Références

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Auteurs

Claire Bar (C)

Department of Paediatric Neurology, Hôpital Necker-Enfants Malades, Reference Centre for Rare Epilepsies, APHP, Bordeaux, France.
Imagine Institute UMR 1163, Paris, France.
Department of Paediatric Neurology, Hôpital des Enfants, CHU Bordeaux, Bordeaux, France.

Jean-Michel Pedespan (JM)

Department of Paediatric Neurology, Hôpital des Enfants, CHU Bordeaux, Bordeaux, France.

Olivia Boccara (O)

Department of Paediatric Dermatology, Hôpital Necker-Enfants Malades, Paris, France.

Nicolas Garcelon (N)

Imagine Institute UMR 1163, Paris, France.
Paris Descartes University, Sorbonne Paris Cite, Paris, France.
Centre de Recherche des Cordeliers, INSERM, UMR 1138 Equipe 22, Paris, France.

Raphael Levy (R)

Department of Paediatric Radiology, Hôpital Necker-Enfants Malades, Paris, France.

David Grévent (D)

Department of Paediatric Radiology, Hôpital Necker-Enfants Malades, Paris, France.

Nathalie Boddaert (N)

Department of Paediatric Radiology, Hôpital Necker-Enfants Malades, Paris, France.

Rima Nabbout (R)

Department of Paediatric Neurology, Hôpital Necker-Enfants Malades, Reference Centre for Rare Epilepsies, APHP, Bordeaux, France.
Imagine Institute UMR 1163, Paris, France.
Paris Descartes University, Sorbonne Paris Cite, Paris, France.

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