Abstract
The authors describe two patients in a Japanese family with autosomal recessive spastic ataxia of Charlevoix-Saguenay. They presented early onset spastic ataxia, sensorimotor neuropathy, nystagmus, slurred speech, and hypermyelinated retinal nerve fibers. The authors identified a homozygous missense mutation (T7492C) in the SACS gene, which resulted in the substitution of arginine for tryptophan at amino acid residue 2498 (W2498R).
Publication types
-
Case Reports
-
Research Support, Non-U.S. Gov't
MeSH terms
-
Adult
-
Ataxia / complications
-
Ataxia / diagnosis
-
Ataxia / genetics*
-
Cerebellum / pathology
-
DNA Mutational Analysis
-
Dysarthria / diagnosis
-
Dysarthria / etiology
-
Dysarthria / genetics
-
Female
-
Gait Disorders, Neurologic / diagnosis
-
Gait Disorders, Neurologic / etiology
-
Gait Disorders, Neurologic / genetics
-
Genes, Recessive
-
Heat-Shock Proteins / genetics*
-
Homozygote
-
Humans
-
Japan
-
Magnetic Resonance Imaging
-
Male
-
Muscle Spasticity / complications
-
Muscle Spasticity / diagnosis
-
Muscle Spasticity / genetics*
-
Mutation, Missense*
-
Nerve Fibers, Myelinated / pathology
-
Neural Conduction
-
Neuropsychological Tests
-
Retina / pathology
-
Siblings
-
Sural Nerve / pathology
-
Sural Nerve / physiopathology
Substances
-
Heat-Shock Proteins
-
SACS protein, human