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Sibling Cases of Charcot-Marie-Tooth Disease Type 4H with a Homozygous FGD4 Mutation and Cauda Equina Thickening.

Authors :
Aoki S
Nagashima K
Shibata M
Kasahara H
Fujita Y
Hashiguchi A
Takashima H
Ikeda Y
Source :
Internal medicine (Tokyo, Japan) [Intern Med] 2021 Dec 15; Vol. 60 (24), pp. 3975-3981. Date of Electronic Publication: 2021 Jun 19.
Publication Year :
2021

Abstract

Charcot-Marie-Tooth disease type 4H (CMT4H) is an autosomal recessive inherited demyelinating neuropathy caused by an FYVE, RhoGEF, and a PH domain-containing protein 4 (FGD4) gene mutation. CMT4H is characterized by an early onset, slow progression, scoliosis, distal muscle atrophy, and foot deformities. We herein present sibling cases of CMT4H with a homozygous mutation in the FGD4 gene. Both patients exhibited cauda equina thickening on magnetic resonance imaging, which had not been reported among the previous CMT4H cases. This is the first report of CMT4H with a homozygous FGD4 c.1730G>A (p.Arg577Gln) mutation showing mild progression and cauda equina thickening.

Details

Language :
English
ISSN :
1349-7235
Volume :
60
Issue :
24
Database :
MEDLINE
Journal :
Internal medicine (Tokyo, Japan)
Publication Type :
Academic Journal
Accession number :
34148957
Full Text :
https://doi.org/10.2169/internalmedicine.7247-21