Literature DB >> 3032454

Cloning and expression of steroid sulfatase cDNA and the frequent occurrence of deletions in STS deficiency: implications for X-Y interchange.

P H Yen, E Allen, B Marsh, T Mohandas, N Wang, R T Taggart, L J Shapiro.   

Abstract

Human STS is a microsomal enzyme important in steroid metabolism. The gene encoding STS is pseudoautosomal in the mouse but not in humans, and escapes X inactivation in both species. We have prepared monoclonal and polyclonal antibodies to the protein which has been purified and from which partial amino acid sequence data have been obtained. cDNA clones containing the entire coding sequence were isolated, sequenced, and expressed in heterologous cells. Variable length transcripts have been shown to be present and due to usage of alternative poly(A) addition sites. The functional gene maps to Xp22.3-Xpter and there is a pseudogene on Yq suggesting a recent pericentric inversion. Absence of STS enzymatic activity occurs frequently in human populations and produces a visible phenotype of scaly skin or ichthyosis. Ten patients with inherited STS deficiency were studied and eight had complete gene deletions. The possibility that STS deficiency results from aberrant X-Y interchange is discussed.

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Year:  1987        PMID: 3032454     DOI: 10.1016/0092-8674(87)90447-8

Source DB:  PubMed          Journal:  Cell        ISSN: 0092-8674            Impact factor:   41.582


  65 in total

1.  Characterization of a low copy repetitive element S232 involved in the generation of frequent deletions of the distal short arm of the human X chromosome.

Authors:  X M Li; P H Yen; L J Shapiro
Journal:  Nucleic Acids Res       Date:  1992-03-11       Impact factor: 16.971

2.  Molecular studies of deletions at the human steroid sulfatase locus.

Authors:  L J Shapiro; P Yen; D Pomerantz; E Martin; L Rolewic; T Mohandas
Journal:  Proc Natl Acad Sci U S A       Date:  1989-11       Impact factor: 11.205

Review 3.  The early and late processing of lysosomal enzymes: proteolysis and compartmentation.

Authors:  A Hasilik
Journal:  Experientia       Date:  1992-02-15

4.  A multipoint linkage map of the distal short arm of the human X chromosome.

Authors:  C L Johnson; P Charmley; P H Yen; L J Shapiro
Journal:  Am J Hum Genet       Date:  1991-08       Impact factor: 11.025

5.  Deletion mapping of 39 random isolated Y-chromosome DNA fragments.

Authors:  C J Oosthuizen; J S Herbert; L K Vermaak; J Brusnicky; J Fricke; L du Plessis; A E Retief
Journal:  Hum Genet       Date:  1990-07       Impact factor: 4.132

6.  Rapid evolution of human pseudoautosomal genes and their mouse homologs.

Authors:  J W Ellison; X Li; U Francke; L J Shapiro
Journal:  Mamm Genome       Date:  1996-01       Impact factor: 2.957

7.  Steroid sulfatase deficiency in Japanese patients: characterization of X-linked ichthyosis by using polymerase chain reaction.

Authors:  T Sugawara; K Honke; S Fujimoto; A Makita
Journal:  Jpn J Hum Genet       Date:  1993-12

8.  Biochemical and immunological characterization of X-linked ichthyosis.

Authors:  X Fan; L Petruschka; K Wulff; U Grimm; F H Herrmann
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

9.  Point mutations in the dystrophin gene.

Authors:  R G Roberts; M Bobrow; D R Bentley
Journal:  Proc Natl Acad Sci U S A       Date:  1992-03-15       Impact factor: 11.205

Review 10.  Androgen synthesis in adrenarche.

Authors:  Walter L Miller
Journal:  Rev Endocr Metab Disord       Date:  2009-03       Impact factor: 6.514

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