Impaired development of melanoblasts in the black-eyed white Mitf(mi-bw) mouse, a model for auditory-pigmentary disorders

Genes to Cells : Devoted to Molecular & Cellular Mechanisms
Hiroki HozumiS Shibahara

Abstract

Microphthalmia-associated transcription factor (Mitf) is a regulator for differentiation of melanoblasts that are derived from the neural crest. The mouse homozygous for the black-eyed white (Mitf(mi-bw)) allele is characterized by the white coat color and deafness, with black eye that is associated with the lack of melanocytes in skin and inner ear. The Mitf(mi-bw) mutation is an insertion of the LINE1 retrotransposable element into intron 3 of the Mitf gene that causes the selective deficiency of the melanocyte-specific Mitf isoform, Mitf-M. Here, we show the expression of Mitf-M mRNA in the trunk region of the homozygous Mitf(mi-bw)(bw) mouse at embryonic days (E) 11.5 and E12.5, but Mitf-M mRNA is undetectable at E13.5. In addition, using bw mouse that carries the lacZ transgene under the control of a melanoblast-specific promoter, we show that the number of migrating melanoblasts in bw embryos was less than 10% of that in control embryos at E11.5 and E12.5, and melanoblasts disappear by E13.5. The loss of melanoblasts in bw embryos was probably caused by apoptosis. Finally, forced expression of Mitf-M in the cultured neural tube of bw embryos ensured the differentiation of melanoblasts. Therefore, the correct dose of Mitf-...Continue Reading

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Citations

Mar 1, 2016·Frontiers in Physiology·Igor Adameyko, Kaj Fried
Nov 3, 2015·Genes to Cells : Devoted to Molecular & Cellular Mechanisms·Koji OhbaShigeki Shibahara
Dec 7, 2013·Genes to Cells : Devoted to Molecular & Cellular Mechanisms·Kazuhisa TakedaShigeki Shibahara
Apr 16, 2013·Seminars in Cell & Developmental Biology·Monika Reissmann, Arne Ludwig
Sep 29, 2019·Pigment Cell & Melanoma Research·Jessica L FlesherAnand K Ganesan
Apr 3, 2018·Genes & Genetic Systems·Hirotoshi ShibuyaHiroaki Yamamoto
Dec 28, 2018·Investigative Ophthalmology & Visual Science·Helen T MichaelChi-Ping Day
Nov 20, 2016·Genes to Cells : Devoted to Molecular & Cellular Mechanisms·Koji OhbaShigeki Shibahara
Aug 11, 2017·The Tohoku Journal of Experimental Medicine·Nattakarn NooronAnchalee Chiabchalard
Jun 27, 2015·Cancer Research·Jenny Li-Ying HuangCatherine D Van Raamsdonk

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Apoptosis

Apoptosis is a specific process that leads to programmed cell death through the activation of an evolutionary conserved intracellular pathway leading to pathognomic cellular changes distinct from cellular necrosis