Literature DB >> 98248

Atypical expression of beta-galactosidase deficiency in a child with Hurler-like features but without neurological abnormalities.

G Andria, E Del Giudice, A J Reuser.   

Abstract

A 28-month-old child was found to have several clinical features of lysosomal storage diseases, including: coarse facies, hepatosplenomegaly, lumbar kyphosis due to hypoplastic beaked L1 and L2 vertebral bodies, vacuolated lymphocytes in blood smears and rare foamy hystiocytes in bone marrow. However, no signs of neurological or ocular abnormalities were detected. A beta-galactosidase deficiency was demonstrated in leukocytes and cultured skin fibroblasts, with a residual activity toward 4-methylumbelliferyl-beta-galactopyranoside ranging between 5 and 15% of the normal mean. Normal activities were found for several other lysosomal acid hydrolases. beta-Galactosidase activities in leukocytes and cultured skin fibroblasts from both parents were within the normal ranges. The patient seems to represent an atypical expression of acid beta-galactosidase deficiency, since his clinical picture does not exaclty correspond to that of either the two classical types of GM1-gangliosidosis or other atypical patients reported in the literature havining beta-galactosidase deficiency.

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Year:  1978        PMID: 98248     DOI: 10.1111/j.1399-0004.1978.tb02055.x

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  9 in total

Review 1.  Biochemistry and genetics of gangliosidoses.

Authors:  K Sandhoff; H Christomanou
Journal:  Hum Genet       Date:  1979       Impact factor: 4.132

2.  Sialidosis and galactosialidosis: chromosomal assignment of two genes associated with neuraminidase-deficiency disorders.

Authors:  O T Mueller; W M Henry; L L Haley; M G Byers; R L Eddy; T B Shows
Journal:  Proc Natl Acad Sci U S A       Date:  1986-03       Impact factor: 11.205

3.  A two-year-old patient with an atypical expression of GM1-beta-galactosidase deficiency: biochemical, immunological, and cell genetic studies.

Authors:  A J Reuser; G Andria; E de Wit-Verbeek; A Hoogeveen; E del Giudice; D Halley
Journal:  Hum Genet       Date:  1979-01-19       Impact factor: 4.132

4.  Molecular defect in combined beta-galactosidase and neuraminidase deficiency in man.

Authors:  A D'Azzo; A Hoogeveen; A J Reuser; D Robinson; H Galjaard
Journal:  Proc Natl Acad Sci U S A       Date:  1982-08       Impact factor: 11.205

5.  The presence of a reduced amount of 32-kd "protective" protein is a distinct biochemical finding in late infantile galactosialidosis.

Authors:  P Strisciuglio; G Parenti; C Giudice; S Lijoi; A T Hoogeveen; A d'Azzo
Journal:  Hum Genet       Date:  1988-11       Impact factor: 4.132

6.  Human beta-galactosidase and alpha-neuraminidase deficient mucolipidosis: genetic complementation analysis of the neuraminidase deficiency.

Authors:  O T Mueller; T B Shows
Journal:  Hum Genet       Date:  1982       Impact factor: 4.132

7.  Turnover of beta-galactosidase in fibroblasts from patients with genetically different types of beta-galactosidase deficiency.

Authors:  O P Van Diggelen; A W Schram; M L Sinnott; P J Smith; D Robinson; H Galjaard
Journal:  Biochem J       Date:  1981-10-15       Impact factor: 3.857

8.  Correction of I-cell defect by hybridization with lysosomal enzyme deficient human fibroblasts.

Authors:  A d'Azzo; D J Halley; A Hoogeveen; H Galjaard
Journal:  Am J Hum Genet       Date:  1980-07       Impact factor: 11.025

9.  A mutation in a mild form of galactosialidosis impairs dimerization of the protective protein and renders it unstable.

Authors:  X Y Zhou; N J Galjart; R Willemsen; N Gillemans; H Galjaard; A d'Azzo
Journal:  EMBO J       Date:  1991-12       Impact factor: 11.598

  9 in total

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