Literature DB >> 6264784

Niemann-Pick disease type B: prenatal diagnosis and enzymatic and chemical studies on fetal brain and liver.

D A Wenger, T Kudoh, M Sattler, M Palmieri, M Yudkoff.   

Abstract

Patients with Niemann-Pick disease type A have a severe neurovisceral disease caused by a deficiency of lysosomal sphingomyelinase activity in all tissues examined. The patients with the type B form have signs and symptoms related to storage of sphingomyelin in the spleen, liver, and lungs, while neurologically they remain normal. They also have a severe deficiency of lysosomal sphingomyelinase activity in all tissues previously examined. Here the brain and liver of a fetus with Niemann-Pick disease type B are examined for enzymatic anc chemical changes. Despite careful analysis, no measurable lysosomal sphingomyelinase could be measured in either organ. Lipid changes were comparable to those observed in fetuses aborted with Niemann-Pick disease type A. The affected child in this family is now age 3 and remains neurologically normal but continues to show organ enlargement and lung infiltration of lipids. It appears that the lack of neurological involvement in type B patients cannot be due to an obvious presence of significant lysosomal sphingomyelinase activity in brain.

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Year:  1981        PMID: 6264784      PMCID: PMC1685041     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  16 in total

1.  Sphingomyelinase isozymes of human tissues: a phpothesis on enzymatic differentiation of the neuropathic and non-neuropathic forms of Niemann-Pick disease.

Authors:  S Yamaguchi; K Suzuki
Journal:  Biochem Biophys Res Commun       Date:  1977-08-08       Impact factor: 3.575

2.  Niemann Pick disease: presence of the magnesium-dependent sphingomyelinase in brain of the infantile form of the disease.

Authors:  S Gatt; T Dinur; J Kopolovic
Journal:  J Neurochem       Date:  1978-08       Impact factor: 5.372

3.  Assay of beta-glucosidase and sphingomyelinase for identification of patients and carriers of Gaucher's and Niemann-Pick diseases.

Authors:  D A Wenger
Journal:  Adv Exp Med Biol       Date:  1978       Impact factor: 2.622

4.  Sphingomyelinase activity at pH 7.4 in human brain and a comparison to activity at pH 5.0.

Authors:  B G Rao; M W Spence
Journal:  J Lipid Res       Date:  1976-09       Impact factor: 5.922

5.  Prenatal Niemann-Pick disease: biochemical and histologic examination of a 19-gestational week fetus.

Authors:  E L Schneider; W G Ellis; R O Brady; J R McCulloch; C J Epstein
Journal:  Pediatr Res       Date:  1972-09       Impact factor: 3.756

6.  I-cell disease: activities of lysosomal enzymes toward natural and synthetic substrates.

Authors:  D A Wenger; M Sattler; C Clark; C Wharton
Journal:  Life Sci       Date:  1976-08-01       Impact factor: 5.037

7.  A practical chromogenic procedure for the detection of homozygotes and heterozygous carriers of Niemann-Pick disease.

Authors:  A E Gal; R O Brady; S R Hibbert; P G Pentchev
Journal:  N Engl J Med       Date:  1975-09-25       Impact factor: 91.245

8.  Chemical and biochemical studies in human fetuses affected with Niemann-Pick disease type A.

Authors:  C Klibansky; S Chazan; A Schoenfeld; A Abramovici
Journal:  Clin Chim Acta       Date:  1979-02-01       Impact factor: 3.786

9.  Prenatal diagnosis and fetal pathology of Niemann-Pick disease.

Authors:  S Higami; K Omura; K Nishizawa; T Yamashita; K Tada
Journal:  Tohoku J Exp Med       Date:  1978-05       Impact factor: 1.848

10.  Niemann-Pick disease (Crocker's group A). Late onset and pigmentary degeneration resembling Hallervorden-Spatz syndrome.

Authors:  J J Martin; M Philippart; J Van Hauwaert; J W Callahan; R Deberdt
Journal:  Arch Neurol       Date:  1972-07
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  5 in total

1.  Interindividual heterogeneity of molecular weight of human brain neutral sphingomyelinase determined by radiation inactivation method.

Authors:  T Levade; R Salvayre; M Potier; L Douste-Blazy
Journal:  Neurochem Res       Date:  1986-08       Impact factor: 3.996

2.  Retroviral-mediated transfer of the human acid sphingomyelinase cDNA: correction of the metabolic defect in cultured Niemann-Pick disease cells.

Authors:  M Suchi; T Dinur; R J Desnick; S Gatt; L Pereira; E Gilboa; E H Schuchman
Journal:  Proc Natl Acad Sci U S A       Date:  1992-04-15       Impact factor: 11.205

3.  Enzyme activities and phospholipid storage patterns in brain and spleen samples from Niemann-Pick disease variants: a comparison of neuropathic and non-neuropathic forms.

Authors:  G T Besley; M Elleder
Journal:  J Inherit Metab Dis       Date:  1986       Impact factor: 4.982

Review 4.  Secondary lipid accumulation in lysosomal disease.

Authors:  Steven U Walkley; Marie T Vanier
Journal:  Biochim Biophys Acta       Date:  2008-12-09

5.  Bone marrow transplantation for Niemann-Pick type IA disease.

Authors:  E Bayever; N Kamani; P Ferreira; G A Machin; M Yudkoff; K Conard; M Palmieri; J Radcliffe; D A Wenger; C S August
Journal:  J Inherit Metab Dis       Date:  1992       Impact factor: 4.982

  5 in total

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