{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T12:26:03Z","timestamp":1790857563365,"version":"4.1.0"},"reference-count":47,"publisher":"The Company of Biologists","issue":"3","license":[{"start":{"date-parts":[[1993,7,1]],"date-time":"1993-07-01T00:00:00Z","timestamp":741484800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[1993,7,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>The receptor tyrosine kinase c-kit and its cognate ligand KL are encoded at the white spotting (W) and steel (Sl) loci of the mouse, respectively. Mutations at both the W and the Sl locus cause deficiencies in gametogenesis, melanogenesis and hematopoiesis (erythrocytes and mast cells). The W-sash mutation differs from most W mutations in that it affects primarily mast cells and melanogenesis but not other cellular targets of W and Sl mutations. Thus, Wsh\/Wsh mice are fertile and not anemic, but they lack mast cells in their skin and intestine and are devoid of coat pigment. Heterozygotes are black with a broad white sash\/belt in the lumbar region. In order to determine the basis for the phenotypes of W-sash mice, we investigated c-kit RNA and protein expression patterns in adult Wsh\/Wsh mice and during embryonic development. We show that c-kit expression is absent in bone-marrow-derived Wsh\/Wsh mast cells, the fetal and the adult lung, and the digestive tract at embryonic day , tissues that normally express c-kit. Unexpectedly, in and Wsh\/Wsh embryos, we found c-kit expression in the dermatome of the somites, the mesenchyme around the otic vesicle and the floorplate of the neural tube, structures known to express the c-kit ligand in wild-type embryos. The ectopic c-kit expression in Wsh homozygous embryos does not affect c-kit ligand expression. The presumed Wsh\/Wsh melanoblasts appeared to be normal and, at similar numbers were found in normal and homozygous mutant embryos. At embryos had a graded distribution of melanoblasts from cranial to caudal with a minimum in the lumbar region. Whereas homozygous Wsh\/Wsh embryos essentially lacked c-kit-positive cells in the skin, heterozygous Wsh\/+ embryos had reduced numbers of melanoblasts compared to +\/+ with few or none in the lumbar region (future sash). It is proposed that ectopic c-kit expression in the somitic dermatome affects early melanogensis in a dominant fashion. Molecular analysis of Wsh chromosomal DNA revealed a deletion or rearrangement in the vicinity of the c-kit gene. These results provide an explanation for the Wsh phenotype and have implications for the control of c-kit expression.<\/jats:p>","DOI":"10.1242\/dev.118.3.705","type":"journal-article","created":{"date-parts":[[2021,4,26]],"date-time":"2021-04-26T01:20:58Z","timestamp":1619400058000},"page":"705-717","source":"Crossref","is-referenced-by-count":102,"title":["<i>W-sash<\/i> affects positive and negative elements controlling c-<i>kit<\/i> expression: ectopic c-<i>kit<\/i> expression at sites of kit-ligand expression affects melanogenesis"],"prefix":"10.1242","volume":"118","author":[{"given":"Regina","family":"Duttlinger","sequence":"first","affiliation":[{"name":"1 Molecular Biology Program and Sloan Kettering Institute and Cornell University, Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"},{"name":"Sloan Kettering Institute and Cornell University 2 Graduate Program in Immunology , , Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katia","family":"Manova","sequence":"additional","affiliation":[{"name":"1 Molecular Biology Program and Sloan Kettering Institute and Cornell University, Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"},{"name":"Cornell University Medical College 3 Department of Cell Biology and Anatomy , , 1300 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tang Y.","family":"Chu","sequence":"additional","affiliation":[{"name":"1 Molecular Biology Program and Sloan Kettering Institute and Cornell University, Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corina","family":"Gyssler","sequence":"additional","affiliation":[{"name":"1 Molecular Biology Program and Sloan Kettering Institute and Cornell University, Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew D.","family":"Zelenetz","sequence":"additional","affiliation":[{"name":"1 Molecular Biology Program and Sloan Kettering Institute and Cornell University, Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rosemary F.","family":"Bachvarova","sequence":"additional","affiliation":[{"name":"Cornell University Medical College 3 Department of Cell Biology and Anatomy , , 1300 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Besmer","sequence":"additional","affiliation":[{"name":"1 Molecular Biology Program and Sloan Kettering Institute and Cornell University, Graduate School of Medical Sciences, 1275 York Avenue, New York, NY 10021, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[1993,7,1]]},"reference":[{"key":"2024121723042244200_DEVELOP_118_3_705C1","doi-asserted-by":"publisher","first-page":"939","DOI":"10.1016\/0955-0674(91)90111-B","article-title":"The kit ligand encoded at the murine steel locus: a pleiotropic growth and differentiation factor","volume":"3","author":"Besmer","year":"1991","journal-title":"Current Opinion in Cell Biology"},{"key":"2024121723042244200_DEVELOP_118_3_705C2","doi-asserted-by":"publisher","first-page":"415","DOI":"10.1038\/320415a0","article-title":"v-kit: oncogene of a new acute transforming feline retrovirus (HZ4-FeSV) - relationship with the protein kinase gene family","volume":"320","author":"Besmer","year":"1986","journal-title":"Nature"},{"key":"2024121723042244200_DEVELOP_118_3_705C3","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1038\/335088a0","article-title":"The proto-oncogene c-kit encoding a transmembrane tyrosine kinase receptor maps to the mouse W locus","volume":"335","author":"Chabot","year":"1988","journal-title":"Nature"},{"key":"2024121723042244200_DEVELOP_118_3_705C4","doi-asserted-by":"publisher","first-page":"523","DOI":"10.1093\/genetics\/23.6.523","article-title":"Studies on spotting patterns. 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