Literature DB >> 15856305

A mutation within the saposin D domain in a Gaucher disease patient with normal glucocerebrosidase activity.

Anna Diaz-Font1, Bru Cormand, Raül Santamaria, Lluïsa Vilageliu, Daniel Grinberg, Amparo Chabás.   

Abstract

Only two Gaucher disease (GD) patients bearing mutations in the prosaposin gene (PSAP), and not in the glucocerebrosidase gene (GBA), have been reported. In both cases, one mutant allele remained unidentified. We report here the identification of the second mutation in one of these patients, being the first complete genotype described so far in a SAP-C-deficient GD patient. This mutation, p.Q430X, is the first one reported in the saposin D domain and probably produces a null allele by nonsense mediated mRNA decay.

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Year:  2005        PMID: 15856305     DOI: 10.1007/s00439-005-1288-x

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  7 in total

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Journal:  Trends Biochem Sci       Date:  1998-06       Impact factor: 13.807

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Journal:  FEBS Lett       Date:  1991-06-17       Impact factor: 4.124

6.  Immunochemical characterization of two activator proteins stimulating enzymic sphingomyelin degradation in vitro. Absence of one of them in a human Gaucher disease variant.

Authors:  H Christomanou; A Aignesberger; R P Linke
Journal:  Biol Chem Hoppe Seyler       Date:  1986-09

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Authors:  M A Rafi; G de Gala; X L Zhang; D A Wenger
Journal:  Somat Cell Mol Genet       Date:  1993-01
  7 in total
  15 in total

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Authors:  Rebecca C Meyer; Michelle M Giddens; Brilee M Coleman; Randy A Hall
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2.  Lack of evidence for genetic association of saposins A, B, C and D with Parkinson's disease.

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Journal:  Brain       Date:  2020-09-01       Impact factor: 13.501

Review 3.  Multi-system disorders of glycosphingolipid and ganglioside metabolism.

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4.  Progranulin mutations result in impaired processing of prosaposin and reduced glucocerebrosidase activity.

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5.  Progranulin-mediated deficiency of cathepsin D results in FTD and NCL-like phenotypes in neurons derived from FTD patients.

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Review 6.  The role of saposin C in Gaucher disease.

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Journal:  Mol Genet Metab       Date:  2012-05-05       Impact factor: 4.797

7.  Sequencing of the GBA coactivator, Saposin C, in Parkinson disease.

Authors:  Bouchra Ouled Amar Bencheikh; Etienne Leveille; Jennifer A Ruskey; Dan Spiegelman; Christopher Liong; Edward A Fon; Guy A Rouleau; Yves Dauvilliers; Nicolas Dupre; Roy N Alcalay; Ziv Gan-Or
Journal:  Neurobiol Aging       Date:  2018-07-02       Impact factor: 4.673

8.  In silico and functional studies of the regulation of the glucocerebrosidase gene.

Authors:  Yotam N Blech-Hermoni; Shira G Ziegler; Kathleen S Hruska; Barbara K Stubblefield; Mary E Lamarca; Matthew E Portnoy; Eric D Green; Ellen Sidransky
Journal:  Mol Genet Metab       Date:  2009-11-04       Impact factor: 4.797

9.  Tissue-specific effects of saposin A and saposin B on glycosphingolipid degradation in mutant mice.

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Review 10.  Glucocerebrosidase is shaking up the synucleinopathies.

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