Three Cases of Hemiconvulsion-Hemiplegia-Epilepsy Syndrome With Focal Cortical Dysplasia Type IIId.

cortical malformation epilepsy surgery focal cortical dysplasia hemiconvulsion-hemiplegia-epilepsy syndrome hemispherotomy histopathology intractable epilepsy pathology

Journal

Frontiers in neurology
ISSN: 1664-2295
Titre abrégé: Front Neurol
Pays: Switzerland
ID NLM: 101546899

Informations de publication

Date de publication:
2019
Historique:
received: 19 08 2019
accepted: 05 11 2019
entrez: 12 12 2019
pubmed: 12 12 2019
medline: 12 12 2019
Statut: epublish

Résumé

Hemiconvulsion-hemiplegia-epilepsy syndrome (HHES) is a subset of acute encephalopathy characterized by infantile-onset with acute hemiconvulsive febrile status and subsequent unilateral cerebral atrophy and hemiparesis. In the chronic phase, patients with HHES develop epilepsy, typically displayed as intractable focal seizures. The patients are often intractable with antiepileptic drugs and need surgical treatment. Although viral encephalitis and genetic abnormalities are presumed to be the underlying etiology, the pathogenesis remains mostly unknown. We describe three cases of successful functional hemispherotomy for intractable epilepsy in HHES. Patients developed acute asymmetrical convulsive status following viral infections during the ages of 17-30 months. Their seizures were intractable with antiepileptic drugs and required hemispherotomy. On the basis of the pathological findings, all cases were diagnosed as focal cortical dysplasia (FCD) type IIId. The epileptogenic mild cortical malformations may be the cause of HHES.

Identifiants

pubmed: 31824410
doi: 10.3389/fneur.2019.01233
pmc: PMC6879674
doi:

Types de publication

Case Reports

Langues

eng

Pagination

1233

Informations de copyright

Copyright © 2019 Itamura, Okanishi, Arai, Nishimura, Baba, Ichikawa, Hirayama, Ishihara, Hiraide, Ishigaki, Fukuda, Otsuki, Enoki and Fujimoto.

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Auteurs

Shinji Itamura (S)

Department of Child Neurology, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Tohru Okanishi (T)

Department of Child Neurology, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Yoshifumi Arai (Y)

Department of Pathology, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Mitsuyo Nishimura (M)

Department of Clinical Laboratory, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Shimpei Baba (S)

Department of Child Neurology, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Naoki Ichikawa (N)

Epilepsy Center, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Yoshimichi Hirayama (Y)

Department of Pediatrics, Naha City Hospital, Okinawa, Japan.

Naoko Ishihara (N)

Department of Pediatrics, Fujita Health University School of Medicine, Toyoake, Japan.

Takuya Hiraide (T)

Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu, Japan.

Hidetoshi Ishigaki (H)

Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu, Japan.

Tokiko Fukuda (T)

Department of Pediatrics, Hamamatsu University School of Medicine, Hamamatsu, Japan.

Yoshiro Otsuki (Y)

Department of Pathology, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Hideo Enoki (H)

Department of Child Neurology, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Ayataka Fujimoto (A)

Epilepsy Center, Seirei-Hamamatsu General Hospital, Shizuoka, Japan.

Classifications MeSH