Document Detail


A case of congenital axonal neuropathy associated with West syndrome.
MedLine Citation:
PMID:  21211919     Owner:  NLM     Status:  Publisher    
Abstract/OtherAbstract:
We report the case of an 11-month-old girl with congenital axonal neuropathy and West syndrome. She had generalized hypotonia and an abnormal posture since birth, and apparently, her development was stalled. Deep tendon reflexes were absent, and at 5months of age, she developed West syndrome followed by refractory seizures. Magnetic resonance imaging of the brain revealed mild cerebral and cerebellar atrophy, high-signal-intensity areas in the white matter, and hypoplasia of the corpus callosum. No action potentials were detected in both lower and upper extremities in motor and sensory conduction velocity analysis performed at 11months of age. Sural nerve biopsy was performed, and analysis of the biopsied specimen revealed axonal degeneration. Originally designed resequencing analysis using microarray was carried out for the 27 genes associated with Charcot-Marie-Tooth disease, but no disease-causing mutations were identified. So far, there have been no reports on simultaneous development of congenital axonal neuropathy and West syndrome.
Authors:
Tomoko Mizuno; Masayuki Sasaki; Hirofumi Komaki; Hiroshi Sakuma; Yoshiaki Saito; Eiji Nakagawa; Kenji Sugai; Yuko Saito; Ikuya Nonaka; Yukio Sawaishi
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Publication Detail:
Type:  JOURNAL ARTICLE     Date:  2011-1-4
Journal Detail:
Title:  Brain & development     Volume:  -     ISSN:  1872-7131     ISO Abbreviation:  -     Publication Date:  2011 Jan 
Date Detail:
Created Date:  2011-1-7     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  7909235     Medline TA:  Brain Dev     Country:  -    
Other Details:
Languages:  ENG     Pagination:  -     Citation Subset:  -    
Copyright Information:
Copyright © 2010 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.
Affiliation:
Department of Child Neurology, National Center Hospital, National Center of Neurology and Psychiatry (NCNP), Tokyo, Japan.
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