Steel-Dickie mutation encodes a c-kit ligand lacking transmembrane and cytoplasmic domains - PubMed (original) (raw)
Steel-Dickie mutation encodes a c-kit ligand lacking transmembrane and cytoplasmic domains
C I Brannan et al. Proc Natl Acad Sci U S A. 1991.
Abstract
Mice homozygous for the viable Sl allele steel-Dickie (Sld) are sterile, severely anemic, and black-eyed white. The nature of the Sld mutation was investigated at the molecular level and was found to be due to a 4.0-kilobase intragenic deletion in mast cell growth factor (MGF) genomic sequences, providing conclusive evidence that Sl encodes MGF. As a consequence of this deletion, Sld is only capable of encoding a soluble truncated growth factor that lacks both transmembrane and cytoplasmic domains. Northern analysis indicates that Sld mRNA is expressed at approximately wild-type levels in adult tissues, and yeast expression studies suggest that the Sld protein is as biologically active as wild-type soluble MGF. These studies provide a molecular basis for explaining the Sld phenotype, a description of a germ-line mutation in the transmembrane and cytoplasmic domains of a membrane-bound growth factor, and in vivo evidence for the importance of membrane-bound forms of growth factors in mammalian development.
Similar articles
- Binding of germ cells to mutant Sld Sertoli cells is defective and is rescued by expression of the transmembrane form of the c-kit ligand.
Marziali G, Lazzaro D, Sorrentino V. Marziali G, et al. Dev Biol. 1993 May;157(1):182-90. doi: 10.1006/dbio.1993.1122. Dev Biol. 1993. PMID: 7683283 - Mast cell growth factor maps near the steel locus on mouse chromosome 10 and is deleted in a number of steel alleles.
Copeland NG, Gilbert DJ, Cho BC, Donovan PJ, Jenkins NA, Cosman D, Anderson D, Lyman SD, Williams DE. Copeland NG, et al. Cell. 1990 Oct 5;63(1):175-83. doi: 10.1016/0092-8674(90)90298-s. Cell. 1990. PMID: 1698554 - Transmembrane form of the kit ligand growth factor is determined by alternative splicing and is missing in the Sld mutant.
Flanagan JG, Chan DC, Leder P. Flanagan JG, et al. Cell. 1991 Mar 8;64(5):1025-35. doi: 10.1016/0092-8674(91)90326-t. Cell. 1991. PMID: 1705866 - The kit-ligand (steel factor) and its receptor c-kit/W: pleiotropic roles in gametogenesis and melanogenesis.
Besmer P, Manova K, Duttlinger R, Huang EJ, Packer A, Gyssler C, Bachvarova RF. Besmer P, et al. Dev Suppl. 1993:125-37. Dev Suppl. 1993. PMID: 7519481 Review. - The kit receptor and its ligand, steel factor, as regulators of hemopoiesis.
Broxmeyer HE, Maze R, Miyazawa K, Carow C, Hendrie PC, Cooper S, Hangoc G, Vadhan-Raj S, Lu L. Broxmeyer HE, et al. Cancer Cells. 1991 Dec;3(12):480-7. Cancer Cells. 1991. PMID: 1726456 Review.
Cited by
- Deranged aortic intima-media thickness, plasma triglycerides and granulopoiesis in Sl/Sl(d) mice.
Dileepan KN, Johnston TP, Li Y, Tawfik O, Stechschulte DJ. Dileepan KN, et al. Mediators Inflamm. 2004 Dec;13(5-6):335-41. doi: 10.1080/09629350400008794. Mediators Inflamm. 2004. PMID: 15770049 Free PMC article. - Expression of functional c-kit receptors rescues the genetic defect of W mutant mast cells.
Alexander WS, Lyman SD, Wagner EF. Alexander WS, et al. EMBO J. 1991 Dec;10(12):3683-91. doi: 10.1002/j.1460-2075.1991.tb04936.x. EMBO J. 1991. PMID: 1718741 Free PMC article. - An allelic series of mutations in the Kit ligand gene of mice. II. Effects of ethylnitrosourea-induced Kitl point mutations on survival and peripheral blood cells of Kitl(Steel) mice.
Rajaraman S, Davis WS, Mahakali-Zama A, Evans HK, Russell LB, Bedell MA. Rajaraman S, et al. Genetics. 2002 Sep;162(1):341-53. doi: 10.1093/genetics/162.1.341. Genetics. 2002. PMID: 12242245 Free PMC article. - Ectodomain shedding of FLT3 ligand is mediated by TNF-alpha converting enzyme.
Horiuchi K, Morioka H, Takaishi H, Akiyama H, Blobel CP, Toyama Y. Horiuchi K, et al. J Immunol. 2009 Jun 15;182(12):7408-14. doi: 10.4049/jimmunol.0801931. J Immunol. 2009. PMID: 19494263 Free PMC article. - Phenotype of White Sika Deer Due to SCF Gene Structural Variation.
Chen X, Dong S, Liu X, Ding N, Xing X. Chen X, et al. Genes (Basel). 2023 May 2;14(5):1035. doi: 10.3390/genes14051035. Genes (Basel). 2023. PMID: 37239395 Free PMC article.
References
- Cell. 1990 Oct 5;63(1):235-43 - PubMed
- Cell. 1990 Oct 5;63(1):167-74 - PubMed
- Cell. 1990 Oct 5;63(1):203-11 - PubMed
- Cell. 1990 Oct 5;63(1):195-201 - PubMed
- Development. 1988 Jan;102(1):117-26 - PubMed
Publication types
MeSH terms
Substances
LinkOut - more resources
Full Text Sources
Molecular Biology Databases
Research Materials