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PMID: 5584 Published · ppublish English Case Reports Journal Article Research Support, U.S. Gov't, Non-P.H.S. Research Support, U.S. Gov't, P.H.S.

Mannosidosis: phenotype of a severely affected child and characterization of alpha-mannosidase activity in cultured fibroblasts from the patient and his parents.

The Journal of pediatrics ·Vol. 88 ·No. 5 ·1976-05-00 ·Pages 814-8

Aylsworth AS, Taylor HA, Stuart CM, Thomas GH

Abstract

A three-year-old boy has coarse facial features, upper respiratory congestion, profound mental retardation, hepatosplenomegaly, increased height and head circumference, cataracts, a gibbus deformity, radiographic changes of dysostosis multiplex, and vacuolized peripheral lymphocytes. These findings are the most commonly reported clinical features in the previously described patients with mannosidosis. Our patient has a severe deficiency, and his parents have intermediate levels, of the acidic component of alpha-mannosidase in their cultured fibroblasts.

MeSH Terms
Cells, Cultured Child, Preschool Disaccharidases/deficiency Face Fibroblasts/enzymology Glycosaminoglycans/urine Hepatomegaly/etiology Humans Hydrogen-Ion Concentration Lysosomes/enzymology Male Mannosidases/deficiency,metabolism Phenotype
Chemicals
Glycosaminoglycans Disaccharidases Mannosidases
Authors & Affiliations
4 authors, click to expand affiliations / ORCID
Aylsworth A S
Taylor H A
Stuart C M
Thomas G H
Article Info
Journal
The Journal of pediatrics
Abbr.
J Pediatr
ISSN
0022-3476
Published
1976-05-00
Pages
814-8
Language
English
Region
United States
NLM ID
0375410
Subset
IM
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