Literature DB >> 2320565

The scurfy mouse mutant has previously unrecognized hematological abnormalities and resembles Wiskott-Aldrich syndrome.

M F Lyon1, J Peters, P H Glenister, S Ball, E Wright.   

Abstract

The X chromosome-linked scurfy (sf) mutant of the mouse is recognized by the scaliness of the skin from which the name is derived and results in death of affected males at about 3-4 weeks of age. Consideration of known man-mouse homologies of the X chromosome prompted hematological studies, which have shown that the blood is highly abnormal. The platelet and erythrocyte counts are both reduced and become progressively lower relative to normal as the disease progresses. There is gastrointestinal bleeding, and most animals appear to die of severe anemia. By contrast, the leukocyte count is consistently raised. Some animals showed signs of infection but it is not yet clear whether there is immunodeficiency. Other features include the scaly skin and apparently reduced lateral growth of the skin, conjunctivitis, and diarrhea in some animals. The mutant resembles Wiskott-Aldrich syndrome in man, which is characterized by thrombocytopenia, eczema, diarrhea, and immunodeficiency. The loci of the human and mouse genes lie in homologous segments of the X chromosome, although apparently in somewhat different positions relative to other gene loci. Scurfy differs from Wiskott-Aldrich syndrome in that scurfy males are consistently hypogonadal.

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Year:  1990        PMID: 2320565      PMCID: PMC53703          DOI: 10.1073/pnas.87.7.2433

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  13 in total

1.  EXCEPTIONAL INHERITANCE OF A SEX-LINKED GENE IN THE MOUSE EXPLAINED ON THE BASIS THAT THE X/O SEX-CHROMOSOME CONSTITUTION IS FEMALE.

Authors:  W L Russell; L B Russell; J S Gower
Journal:  Proc Natl Acad Sci U S A       Date:  1959-04       Impact factor: 11.205

2.  Multilocus molecular mapping of the mouse X chromosome.

Authors:  L J Mullins; S G Grant; D A Stephenson; V M Chapman
Journal:  Genomics       Date:  1988-10       Impact factor: 5.736

3.  Hereditary X-linked thrombocytopenia maps to the same chromosomal region as the Wiskott-Aldrich syndrome.

Authors:  M Donnér; M Schwartz; K U Carlsson; L Holmberg
Journal:  Blood       Date:  1988-12       Impact factor: 22.113

4.  Linkage of the Wiskott-Aldrich syndrome with polymorphic DNA sequences from the human X chromosome.

Authors:  M Peacocke; K A Siminovitch
Journal:  Proc Natl Acad Sci U S A       Date:  1987-05       Impact factor: 11.205

5.  Report of the committee on the genetic constitution of the X chromosome.

Authors:  J L Mandel; H F Willard; R L Nussbaum; K E Davies; G Romeo
Journal:  Cytogenet Cell Genet       Date:  1988

6.  Genetic control over the inactivation of autosomal genes attached to the X-chromosome.

Authors:  B M Cattanach; J H Isaacson
Journal:  Z Vererbungsl       Date:  1965

7.  Conservation and reorganization of loci on the mammalian X chromosome: a molecular framework for the identification of homologous subchromosomal regions in man and mouse.

Authors:  L C Amar; L Dandolo; A Hanauer; A R Cook; D Arnaud; J L Mandel; P Avner
Journal:  Genomics       Date:  1988-04       Impact factor: 5.736

Review 8.  Chromosome maps of man and mouse. IV.

Authors:  A G Searle; J Peters; M F Lyon; J G Hall; E P Evans; J H Edwards; V J Buckle
Journal:  Ann Hum Genet       Date:  1989-05       Impact factor: 1.670

9.  Expression on blood cells of sialophorin, the surface glycoprotein that is defective in Wiskott-Aldrich syndrome.

Authors:  E Remold-O'Donnell; C Zimmerman; D Kenney; F S Rosen
Journal:  Blood       Date:  1987-07       Impact factor: 22.113

Review 10.  Wiskott-Aldrich syndrome: new perspectives in pathogenesis and management.

Authors:  G R Standen
Journal:  J R Coll Physicians Lond       Date:  1988-04
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  63 in total

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Authors:  S D Brown; P Avner; G E Herman
Journal:  Mamm Genome       Date:  1992       Impact factor: 2.957

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Authors:  D D Patel
Journal:  J Clin Invest       Date:  2001-01       Impact factor: 14.808

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Review 5.  Mouse X chromosome.

Authors:  S D Brown; P Avner; V M Chapman; R M Hamvas; G E Herman
Journal:  Mamm Genome       Date:  1991       Impact factor: 2.957

Review 6.  Therapeutic targeting of Janus kinases.

Authors:  Marko Pesu; Arian Laurence; Nandini Kishore; Samuel H Zwillich; Gary Chan; John J O'Shea
Journal:  Immunol Rev       Date:  2008-06       Impact factor: 12.988

Review 7.  Fetal regulatory T cells and peripheral immune tolerance in utero: implications for development and disease.

Authors:  Trevor D Burt
Journal:  Am J Reprod Immunol       Date:  2013-02-25       Impact factor: 3.886

8.  Circulating integrin alpha4/beta7+ lymphocytes targeted by vedolizumab have a pro-inflammatory phenotype.

Authors:  James D Lord; S Alice Long; Donna M Shows; Jerill Thorpe; Katherine Schwedhelm; Janice Chen; Mariko Kita; Jane H Buckner
Journal:  Clin Immunol       Date:  2018-05-26       Impact factor: 3.969

Review 9.  Clinical and molecular features of the immunodysregulation, polyendocrinopathy, enteropathy, X linked (IPEX) syndrome.

Authors:  R S Wildin; S Smyk-Pearson; A H Filipovich
Journal:  J Med Genet       Date:  2002-08       Impact factor: 6.318

10.  Induction of FoxP3 and acquisition of T regulatory activity by stimulated human CD4+CD25- T cells.

Authors:  Mindi R Walker; Deborah J Kasprowicz; Vivian H Gersuk; Angele Benard; Megan Van Landeghen; Jane H Buckner; Steven F Ziegler
Journal:  J Clin Invest       Date:  2003-11       Impact factor: 14.808

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