Juvenile GM<sub>1</sub> gangliosidosis: Clinical, pathological, chemical and enzymatic studies

J S O′Brien(University of California San Diego), M. W. Ho(University of California San Diego), M. Lois Veath(University of California San Diego), John F. Wilson(Primary Children's Hospital), Garth G. Myers(Primary Children's Hospital), John M. Opitz(March of Dimes), G. M. ZuRhein(Oklahoma State University Center for Health Sciences), Jürgen W. Spranger(Universitäts-Kinderklinik Würzburg), Henrik Hartmann(Oklahoma State University Center for Health Sciences), Bjørn Haneberg(Haukeland University Hospital), F. R. Grosses(Universitäts-Kinderklinik Würzburg)
Clinical Genetics
November 1, 1972
Cited by 47

Abstract

Clinical, histological, chemical and enzymatic studies are reported in five patients in two families with juvenile GM 1 gangliosidosis (GM 1 gangliosidosis, Type 2). The clinical picture includes onset of mental and motor retardation beginning at about one year of age and progressive neurological decline with death usually within the first decade. Bony deformities, although mild, are often present and are diagnostically helpful. Ganglioside GM 1 and a galactose‐containing polysaccharide (similar to keratan sulfate) are stored, β‐galactosidase activities are reduced in tissues and cultured skin fibroblasts; both homozygotes and heterozygotes can be detected by this method. The differentiation of juvenile GM 1 gangliosidosis from generalized gangliosidosis (GM 1 gangliosidosis, Type 1) is discussed.


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