Brannan C I, Lyman S D, Williams D E, Eisenman J, Anderson D M, Cosman D, Bedell M A, Jenkins N A, Copeland N G
Mammalian Genetics Laboratory, National Cancer Institute-Frederick Cancer Research and Development Center, MD 21702-1201.
Proc Natl Acad Sci U S A. 1991 Jun 1;88(11):4671-4. doi: 10.1073/pnas.88.11.4671.
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.