Identification of kit-ligand a as the gene responsible for the medaka pigment cell mutant few melanophore

Yuji Otsuki, Yuki Okuda, Kiyoshi Naruse, Hideyuki Saya

Research output: Contribution to journalArticle

Abstract

The body coloration of animals is due to pigment cells derived from neural crest cells, which are multipotent and differentiate into diverse cell types. Medaka (Oryzias latipes) possesses four distinct types of pigment cells known as melanophores, xanthophores, iridophores, and leucophores. The few melanophore (fm) mutant of medaka is characterized by reduced numbers of melanophores and leucophores. We here identify kit-ligand a (kitlga) as the gene whose mutation gives rise to the fm phenotype. This identification was confirmed by generation of kitlga knockout medaka and the findings that these fish also manifest reduced numbers of melanophores and leucophores and fail to rescue the fm mutant phenotype. We also found that expression of sox5, pax7a, pax3a, and mitfa genes is down-regulated in both fm and kitlga knockout medaka, implicating c-Kit signaling in regulation of the expression of these genes as well as the encoded transcription factors in pigment cell specification. Our results may provide insight into the pathogenesis of c-Kit–related pigmentation disorders such as piebaldism in humans, and our kitlga knockout medaka may prove useful as a tool for drug screening.

Original languageEnglish
Pages (from-to)311-319
Number of pages9
JournalG3: Genes, Genomes, Genetics
Volume10
Issue number1
DOIs
Publication statusPublished - 2020 Jan 1

Fingerprint

Melanophores
Oryzias
Stem Cell Factor
Genes
Piebaldism
Pigmentation Disorders
Phenotype
Preclinical Drug Evaluations
Neural Crest
Gene Expression Regulation
Fishes
Transcription Factors
Mutation

Keywords

  • Few melanophore mutant
  • Kit-ligand
  • Leucophore
  • Medaka
  • Melanophore

ASJC Scopus subject areas

  • Molecular Biology
  • Genetics
  • Genetics(clinical)

Cite this

Identification of kit-ligand a as the gene responsible for the medaka pigment cell mutant few melanophore. / Otsuki, Yuji; Okuda, Yuki; Naruse, Kiyoshi; Saya, Hideyuki.

In: G3: Genes, Genomes, Genetics, Vol. 10, No. 1, 01.01.2020, p. 311-319.

Research output: Contribution to journalArticle

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