Chronic subdural hematoma: A previously unreported life-threatening complication in adult with Sotos syndrome.


Journal

American journal of medical genetics. Part A
ISSN: 1552-4833
Titre abrégé: Am J Med Genet A
Pays: United States
ID NLM: 101235741

Informations de publication

Date de publication:
12 2020
Historique:
received: 25 06 2020
revised: 28 08 2020
accepted: 02 09 2020
pubmed: 15 10 2020
medline: 25 6 2021
entrez: 14 10 2020
Statut: ppublish

Résumé

Macrocephaly, defined as head circumference ≥ 2 SDs, is a cardinal feature of Sotos syndrome (SS) and generally persists in adulthood. Subdural fluid collection, typically associated with macrocephaly, is described in children due to anatomical conformation, and in adulthood due to brain atrophy and ex-vacuo hydrocephalus. On the other hand, a true, symptomatic, chronic subdural hematoma (CSH) is a previously unreported complication of SS in adulthood. Here we describe the first SS patient presenting symptomatic CSH, leading to frequent hospitalizations for surgical evacuations that consistently recurred. Middle meningeal artery (MMA) embolization and epidural blood patch (EBP) allowed to resolve the CSH with complete resolution of clinical signs and symptoms. We hypothesize that appearance and recurrences of CSH may be related to pathological biomechanics of brain, cerebro-spinal fluid and skull, secondary to anatomical features of SS. In this context, surgical evacuation may be less efficient than usual to cure CSH. Alternative treatment to avoid blood extravasation, as MMA embolization, or to cure concurrent causes of the pathology, as EBP, may be considered.

Identifiants

pubmed: 33052010
doi: 10.1002/ajmg.a.61900
doi:

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

3052-3055

Informations de copyright

© 2020 Wiley Periodicals LLC.

Références

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Auteurs

Diana Carli (D)

Department of Public Health and Pediatric Sciences, School of Medicine, University of Torino, Torino, Italy.

Andrea Gazzin (A)

Department of Public Health and Pediatric Sciences, School of Medicine, University of Torino, Torino, Italy.

Maria Roberta Bongioanni (MR)

Department of Neurology, Santissima Annunziata Hospital, Savigliano, Italy.

Mauro Bergui (M)

Department of Neuroscience, Neuroradiological Unit, University of Torino, Azienda Ospedaliera Città della Salute e della Scienza Hospital, Torino, Italy.

Alessandro Mussa (A)

Department of Public Health and Pediatric Sciences, School of Medicine, University of Torino, Torino, Italy.

Giovanni Battista Ferrero (GB)

Department of Public Health and Pediatric Sciences, School of Medicine, University of Torino, Torino, Italy.
Department of Clinical and Biological Sciences, School of Medicine, University of Torino, Torino, Italy.

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