Literature DB >> 8493781

Fetal Niemann-Pick disease type C: ultrastructural and lipid findings in liver and spleen.

C Dumontel1, C Girod, F Dijoud, Y Dumez, M T Vanier.   

Abstract

We present the first ultrastructural study of liver and spleen from a 20-week fetus with Niemann-Pick disease type C in correlation with lipid studies of these tissues. The lipid storage pattern was characteristic of the disease and although the distribution of the lipid storage was similar to that of affected children, ultrastructural studies emphasized that many inclusions were qualitatively different. These are discussed. Concomitant with this complex lipid storage, ultrastructural evidence of cholestasis was observed and the early hyperplasia of pericanalicular microfilaments leads us to question the presence of a toxic metabolite which might induce cholestasis by acting upon microfilaments.

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Year:  1993        PMID: 8493781     DOI: 10.1007/BF01621810

Source DB:  PubMed          Journal:  Virchows Arch A Pathol Anat Histopathol        ISSN: 0174-7398


  41 in total

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Journal:  Acta Neuropathol       Date:  1978-06-30       Impact factor: 17.088

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Authors:  G Adam; R J Brereton; M Agrawal; B D Lake
Journal:  J Pediatr Gastroenterol Nutr       Date:  1988 Jan-Feb       Impact factor: 2.839

6.  Niemann-Pick disease type B: first-trimester prenatal diagnosis on chorionic villi and biochemical study of a foetus at 12 weeks of development.

Authors:  M T Vanier; J Boue; Y Dumez
Journal:  Clin Genet       Date:  1985-10       Impact factor: 4.438

7.  Niemann-Pick type C disease and early cholestasis in three brothers.

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Journal:  Acta Paediatr Belg       Date:  1980 Jan-Mar

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Journal:  Ann Pathol       Date:  1989       Impact factor: 0.407

9.  Niemann-Pick disease type C. Pathological, histochemical, ultrastructural and biochemical studies.

Authors:  E F Gilbert; J Callahan; C Viseskul; J M Opitz
Journal:  Eur J Pediatr       Date:  1981-07       Impact factor: 3.183

10.  Familial cholestatic cirrhosis associated with Kayser-Fleischer rings.

Authors:  C Kaplinsky; I Sternlieb; N Javitt; Y Rotem
Journal:  Pediatrics       Date:  1980-04       Impact factor: 7.124

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  8 in total

Review 1.  Lipid changes in Niemann-Pick disease type C brain: personal experience and review of the literature.

Authors:  M T Vanier
Journal:  Neurochem Res       Date:  1999-04       Impact factor: 3.996

2.  Recovery from liver disease in a Niemann-Pick type C mouse model.

Authors:  Naomi L Sayre; Victoria M Rimkunas; Mark J Graham; Rosanne M Crooke; Laura Liscum
Journal:  J Lipid Res       Date:  2010-04-24       Impact factor: 5.922

3.  TNF-{alpha} plays a role in hepatocyte apoptosis in Niemann-Pick type C liver disease.

Authors:  Victoria M Rimkunas; Mark J Graham; Rosanne M Crooke; Laura Liscum
Journal:  J Lipid Res       Date:  2008-09-24       Impact factor: 5.922

4.  Ultrastructural changes in the lung in Niemann-Pick type C mouse.

Authors:  T Manabe; T Yamane; Y Higashi; P G Pentchev; K Suzuki
Journal:  Virchows Arch       Date:  1995       Impact factor: 4.064

5.  Cholesterol and oxygenated cholesterol concentrations are markedly elevated in peripheral tissue but not in brain from mice with the Niemann-Pick type C phenotype.

Authors:  G S Tint; P Pentchev; G Xu; A K Batta; S Shefer; G Salen; A Honda
Journal:  J Inherit Metab Dis       Date:  1998-12       Impact factor: 4.982

6.  In vivo antisense oligonucleotide reduction of NPC1 expression as a novel mouse model for Niemann Pick type C- associated liver disease.

Authors:  Victoria M Rimkunas; Mark J Graham; Rosanne M Crooke; Laura Liscum
Journal:  Hepatology       Date:  2008-05       Impact factor: 17.425

Review 7.  Lysosomal storage diseases.

Authors:  Carlos R Ferreira; William A Gahl
Journal:  Transl Sci Rare Dis       Date:  2017-05-25

Review 8.  Current Challenges in Understanding the Cellular and Molecular Mechanisms in Niemann-Pick Disease Type C1.

Authors:  Anja U Bräuer; Angela Kuhla; Carsten Holzmann; Andreas Wree; Martin Witt
Journal:  Int J Mol Sci       Date:  2019-09-06       Impact factor: 5.923

  8 in total

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