Literature DB >> 6422434

Complementation, cross correction, and drug correction studies of combined beta-galactosidase neuraminidase deficiency in human fibroblasts.

P Strisciuglio, K E Creek, W S Sly.   

Abstract

Neuraminidase activity in fibroblasts obtained from a patient with combined beta-galactosidase-neuraminidase deficiency (beta-gal-/neur-) was partially restored by fusion with two ML I cell lines and an ML II cell line. As observed with neuraminidase activity, beta-galactosidase also showed complementation with an increase in activity when beta-gal-/neur- fibroblasts were fused with an ML II or a GMI gangliosidosis cell line. Both GMI gangliosidosis and sialidosis fibroblasts secreted a "corrective factor" which, when added to medium above beta-gal-/neur- fibroblasts, was pinocytosed and partially corrected its deficiencies for these two enzymes. This partial correction of beta-galactosidase and neuraminidase activities persisted for at least 72 h after removal of the "corrective factor" from the medium. A "corrective factor" with similar properties was obtained from glycoproteins isolated by chromatography of human spleen homogenates on concanavalin A-Sepharose. Treatment of beta-gal-/neur- fibroblasts with leupeptin or EP475, two inhibitors of lysosomal thiol-proteases, partially restored beta-galactosidase activity but caused no significant improvement in neuraminidase levels. The partial corrective effect of leupeptin on beta-galactosidase activity persisted for at least 2 d after removal of the drug, even in the presence of cycloheximide.

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Year:  1984        PMID: 6422434     DOI: 10.1203/00006450-198402000-00011

Source DB:  PubMed          Journal:  Pediatr Res        ISSN: 0031-3998            Impact factor:   3.756


  6 in total

1.  Effects of irradiation on the postnatal development of the brain in a genetic mouse model of globoid cell leukodystrophy.

Authors:  Francesca Galbiati; Giulia Clementi; Daniela Superchi; Maria I Givogri; Ernesto R Bongarzone
Journal:  Neurochem Res       Date:  2007-01-03       Impact factor: 3.996

2.  Galactosialidosis: molecular heterogeneity among distinct clinical phenotypes.

Authors:  S Palmeri; A T Hoogeveen; F W Verheijen; H Galjaard
Journal:  Am J Hum Genet       Date:  1986-02       Impact factor: 11.025

3.  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

4.  Biochemical phenotyping of a single sibship with both cystinosis and Fabry disease.

Authors:  W A Gahl; M Adamson; I Kaiser-Kupfer; I H Ludwig; H J O'Connell; W Cohen; J Barranger
Journal:  J Inherit Metab Dis       Date:  1985       Impact factor: 4.982

5.  Overexpression of V-ATPase B2 attenuates lung injury/fibrosis by stabilizing lysosomal membrane permeabilization and increasing collagen degradation.

Authors:  Jong-Uk Lee; Jisu Hong; Hyesun Shin; Chnag-Beom Ryu; Sung-Woo Park; Sung Hwan Jeong
Journal:  Exp Mol Med       Date:  2022-05-27       Impact factor: 12.153

6.  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 in total

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