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PMID: 408464 Published · ppublish English Case Reports Journal Article

Juvenile Gaucher's disease with horizontal gaze palsy in three siblings.

Journal of neurology, neurosurgery, and psychiatry ·Vol. 40 ·No. 5 ·1977-05-00 ·Pages 470-8

Tripp JH, Lake BD, Young E, Ngu J, Brett EM

Abstract

Three children in a West African family had Gaucher's disease of juvenile onset (Type 3), and all showed an identical neurological disorder. The diagnosis was substantiated by histochemical demonstration of Gaucher cells in bone marrow, liver, and spleen, the finding of an excess of glucosyl ceramides in a liver extract, and a deficient activity of the enzyme beta-glucosidase in cultured skin fibroblasts. The neurological picture was characterised by myoclonic epilepsy, muscle wasting, hypotonia, pyramidal signs, some intellectual deterioration, and a striking disturbance of eye movements. The latter appears to result from specific involvement of the supranuclear pathways subserving lateral gaze. The distinctive features of this clinical syndrome are emphasised.

MeSH Terms
Age Factors Bone Marrow/pathology Child Electroencephalography Epilepsies, Myoclonic/complications Female Gaucher Disease/complications,genetics,pathology,physiopathology Humans Liver/ultrastructure Male Ophthalmoplegia/complications,genetics,physiopathology Pedigree Syndrome
Authors & Affiliations
5 authors, click to expand affiliations / ORCID
Tripp J H
Lake B D
Young E
Ngu J
Brett E M
References (13)
13 references, click to expand
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Article Info
Journal
Journal of neurology, neurosurgery, and psychiatry
Abbr.
J Neurol Neurosurg Psychiatry
ISSN
0022-3050
Published
1977-05-00
Pages
470-8
Language
English
Region
England
NLM ID
2985191R
PMCID
PMC492722
Subset
IM
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