Literature DB >> 3864728

Recessive X-linked ichthyosis: lack of immunologically detectable steroid sulfatase enzyme protein.

E H Epstein, J M Bonifas.   

Abstract

Patients with recessive X-linked ichthyosis (RXLI), one hereditary form of scaly skin, lack activity of the enzyme steroid sulfatase in all tissues studied. To investigate the molecular defect underlying the lack of enzyme activity, we prepared antisera against normal enzyme by injecting normal placental microsomal suspensions or partially purified steroid sulfatase into rabbits. Antibody activity was assessed by immunoprecipitation of detergent solubilized steroid sulfatase. In addition, we prepared rabbit antisera against RXLI placental microsomal suspensions. To detect immunologically cross-reactive material in patients' placentas, extracts were studied by immunoblot techniques and by competition with normal enzyme for antibody binding. Patients' extracts did not contain immunoreactive material co-migrating on electrophoresis with purified enzyme nor did they inhibit immunoprecipitation of normal enzyme. Sera from rabbits immunized with RXLI placental microsomes contain no antibodies to normal steroid sulfatase, as judged by their failure to immunoprecipitate normal enzyme or to react with normal steroid sulfatase on immunoblot. Thus the mutation in RXLI appears to reduce steroid sulfatase enzyme protein as well as enzyme activity.

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Year:  1985        PMID: 3864728     DOI: 10.1007/bf00284573

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


  24 in total

Review 1.  Mutation and enzyme function in humans.

Authors:  H E Sutton; R P Wagner
Journal:  Annu Rev Genet       Date:  1975       Impact factor: 16.830

2.  Steroid sulfatase deficiency.

Authors:  L J Shapiro; L Cousins; A L Fluharty; R L Stevens; H Kihara
Journal:  Pediatr Res       Date:  1977-08       Impact factor: 3.756

3.  Cleavage of structural proteins during the assembly of the head of bacteriophage T4.

Authors:  U K Laemmli
Journal:  Nature       Date:  1970-08-15       Impact factor: 49.962

4.  Deficiency of arylsulfatase C in cultured skin fibroblasts of X-linked ichthyosis.

Authors:  J C Meyer; H Weiss; H P Grundmann; T G Würsch; U W Schnyder
Journal:  Hum Genet       Date:  1979       Impact factor: 4.132

5.  Studies of the biochemical basis of steroid sulphatase deficiency: preliminary evidence suggesting a defect in membrane-enzyme structure.

Authors:  R W McNaught; J T France
Journal:  J Steroid Biochem       Date:  1980-03       Impact factor: 4.292

6.  Is multiple sulphatase deficiency due to defective regulation of sulphohydrolase expression?

Authors:  M B Fiddler; D Vine; E Shapira; H L Nadler
Journal:  Nature       Date:  1979-11-01       Impact factor: 49.962

7.  Rapid laboratory diagnostic of X-linked ichthyosis.

Authors:  J C Meyer; V Groh; V Giger; H Weiss; H Varbelow; U W Schnyder
Journal:  Dermatologica       Date:  1982-04

8.  Human placental steroid sulfatase: purification and properties.

Authors:  H Noël; L Plante; G Bleau; A Chapdelaine; K D Roberts
Journal:  J Steroid Biochem       Date:  1983-11       Impact factor: 4.292

9.  X-linked ichthyosis: increased blood cholesterol sulfate and electrophoretic mobility of low-density lipoprotein.

Authors:  E H Epstein; R M Krauss; C H Shackleton
Journal:  Science       Date:  1981-11-06       Impact factor: 47.728

10.  X-linked steroid sulfatase: evidence for different gene-dosage in males and females.

Authors:  C R Müller; B Migl; H Traupe; H H Ropers
Journal:  Hum Genet       Date:  1980       Impact factor: 4.132

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  6 in total

1.  Isolation and characterization of a steroid sulfatase cDNA clone: genomic deletions in patients with X-chromosome-linked ichthyosis.

Authors:  A Ballabio; G Parenti; R Carrozzo; G Sebastio; G Andria; V Buckle; N Fraser; I Craig; M Rocchi; G Romeo
Journal:  Proc Natl Acad Sci U S A       Date:  1987-07       Impact factor: 11.205

2.  Unique alpha-spectrin mutant in a kindred with common hereditary elliptocytosis.

Authors:  P A Lane; R L Shew; T A Iarocci; N Mohandas; T Hays; W C Mentzer
Journal:  J Clin Invest       Date:  1987-03       Impact factor: 14.808

3.  Studies on cross-reacting material to steroid sulphatase in fibroblasts from patients affected by different types of steroid sulphatase deficiency.

Authors:  G Parenti; A Ballabio; A T Hoogeveen; C M Van der Loos; A C Jobsis; G Andria
Journal:  J Inherit Metab Dis       Date:  1987       Impact factor: 4.982

4.  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

5.  Deletions of the steroid sulphatase gene in "classical" X-linked ichthyosis and in X-linked ichthyosis associated with Kallmann syndrome.

Authors:  A Ballabio; G Sebastio; R Carrozzo; G Parenti; A Piccirillo; M G Persico; G Andria
Journal:  Hum Genet       Date:  1987-12       Impact factor: 4.132

6.  Cloning of a cDNA for steroid sulfatase: frequent occurrence of gene deletions in patients with recessive X chromosome-linked ichthyosis.

Authors:  J M Bonifas; B J Morley; R E Oakey; Y W Kan; E H Epstein
Journal:  Proc Natl Acad Sci U S A       Date:  1987-12       Impact factor: 11.205

  6 in total

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