Transient hypoglycorrhachia with paroxysmal abnormal eye movement in early infancy.

Abnormal eye movement Early infancy Glucose transporter 1 Hypoglycorrhachia

Journal

Brain & development
ISSN: 1872-7131
Titre abrégé: Brain Dev
Pays: Netherlands
ID NLM: 7909235

Informations de publication

Date de publication:
Mar 2021
Historique:
received: 24 06 2020
revised: 30 10 2020
accepted: 08 11 2020
pubmed: 30 11 2020
medline: 5 10 2021
entrez: 29 11 2020
Statut: ppublish

Résumé

Paroxysmal abnormal eye movement in early infancy is one of the initial symptoms of glucose transporter 1 deficiency syndrome (GLUT1DS). We describe four early infants with transient hypoglycorrhachia presenting with abnormal eye movements. Their symptoms disappeared after the introduction of a ketogenic diet (KD), and their development was normal. Since no variants in SLC2A1 were detected, the CSF-to-blood glucose ratios (C/B) were re-examined, and within normal range. None of the four patients displayed recurrent symptoms after withdrawal from the KD. Because long-term KD has potential adverse effects and could affect the quality of life of patients and their families, re-examination of CSF glucose during late infancy should be considered in the case of absence of the SLC2A1 pathogenic variant.

Identifiants

pubmed: 33248857
pii: S0387-7604(20)30317-X
doi: 10.1016/j.braindev.2020.11.006
pii:
doi:

Substances chimiques

Glucose IY9XDZ35W2

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

482-485

Informations de copyright

Copyright © 2020 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Auteurs

Daisuke Tajima (D)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan; Department of Pediatrics, Karatsu Red Cross Hospital, Japan. Electronic address: daisuke-tajima@karatsu.jrc.or.jp.

Takuji Nakamura (T)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan.

Fumio Ichinose (F)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan.

Nobuhiko Okamoto (N)

Department of Medical Genetics, Osaka Women's and Children's Hospital, Japan.

Yuko Tomonoh (Y)

Department of Pediatrics, Faculty of Medicine, Fukuoka University, Japan.

Keiko Uda (K)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan.

Rie Furukawa (R)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan; Department of Pediatrics, Karatsu Red Cross Hospital, Japan.

Katsuya Tashiro (K)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan; Department of Pediatrics, Karatsu Red Cross Hospital, Japan.

Muneaki Matsuo (M)

Department of Pediatrics, Faculty of Medicine, Saga University, Japan.

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Classifications MeSH