FOXD3 regulates the lineage switch between neural crest-derived glial cells and pigment cells by repressing MITF through a non-canonical mechanism
Open accessAbstract
The first neural crest cells to emigrate from the neural tube are specified as neurons and glial cells and are subsequently followed by melanocytes of the skin. We wished to understand how this fate switch is controlled. The transcriptional repressor FOXD3 is expressed exclusively in the neural/glial precursors and MITF is expressed only in melanoblasts. Moreover, FOXD3 represses melanogenesis. Here we show that avian MITF expression begins very early during melanoblast migration and that loss of MITF in melanoblasts causes them to transdifferentiate to a glial phenotype. Ectopic expression of FOXD3 represses MITF in cultured neural crest cells and in B16-F10 melanoma cells. We also show that FOXD3 does not bind directly to the MITFpromoter, but instead interacts with the transcriptional activator PAX3 to prevent the binding of PAX3 to the MITF promoter. Overexpression of PAX3 is sufficient to rescue MITF expression from FOXD3-mediated repression. We conclude that FOXD3 controls the lineage choice between neural/glial and pigment cells by repressing MITF during the early phase of neural crest migration.
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Foxd3 maintains pluripotency of neural crest cells and later contributes to specifying neuronal, glial, and iridophore fates over melanocyte or melanophore fates.
“Foxd3 maintains pluripotency of neural crest cells and later contributes to specifying neuronal, glial, and iridophore fates over melanocyte or melanophore fates (51, 52, 60) and is regulated by BMP signals in other contexts (61, 62).”
From Agouti and BMP signaling drive a naturally occurring fate conversion of melanophores to leucophores in zebrafish (2025) · cites this paper as [51]
Foxd3 has been proposed to repress mitfa expression, biasing bipotent melanoiridoblast progenitors towards an iridophore fate.
“the Forkhead box D3 transcription factor (Foxd3) has been proposed to repress mitfa expression, consistent with the suggested role of FOXD3 in repressing MITF in other models [30], [31], thus biasing bipotent melanoiridoblast progenitors towards an iridophore fate [15], [32].”
From A systems biology approach uncovers the core gene regulatory network governing iridophore fate choice from the neural crest (2018) · cites this paper as [31]
Cites
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Wilkins, A. S., Wrangham, R. W., & Fitch, W. T. (2014) · Genetics · doi:10.1534/genetics.114.165423 · Cited by 751 · see citing works
The melanocyte lineage in development and disease
Mort, R. L., Jackson, I. J., & Patton, E. E. (2015) · Development · doi:10.1242/dev.106567 · Cited by 416 · see citing works
Mutations in MITF and PAX3 cause “splashed white” and other white spotting phenotypes in horses
Hauswirth, R., Haase, B., Blatter, M., Brooks, S. A., Burger, D., Drögemüller, C., Gerber, V., Henke, D., Janda, J., Jude, R., et al. (2012) · PLoS Genetics · doi:10.1371/journal.pgen.1002653 · Cited by 170 · see citing works
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PAX3 and FOXD3 Promote CXCR4 Expression in Melanoma
Kubic, J. D., Lui, J. W., Little, E. C., Ludvik, A. E., Konda, S., Salgia, R., Aplin, A. E., & Lang, D. (2015) · Journal of Biological Chemistry · doi:10.1074/jbc.m115.670976 · Cited by 25 · see citing works
Huang, D., Kapadia, E. H., Liang, Y., Shriver, L. P., Dai, S., Patti, G. J., Humbel, B. M., Laudet, V., & Parichy, D. M. (2025) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.2424180122 · Cited by 15 · see citing works
- Authors
- Thomas, A. J., & Erickson, C. A.
- Affiliations
- University of California, Davis (US)
- ORCID
- Thomas, Aaron J.
- Venue
- Development
- Publisher
- The Company of Biologists
- Published
- 2009-06-01
- Volume
- 136
- Issue
- 11
- Pages
- 1849-1858
- ISSN
- 0950-1991 (ISSN-L)
- 1477-9129
- PMC
- PMC2680109
- OpenAlex
- W1986238441
- Cited by
- 176
- References
- 1
- Citations by year
- 2026: 5 · 2025: 8 · 2024: 6 · 2023: 4 · 2022: 5 · 2021: 14 · 2020: 8 · 2019: 10 · 2018: 7 · 2017: 14 · 2016: 10 · 2015: 16 · 2014: 8 · 2013: 18 · 2012: 12
- FWCI
- 5.94
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Locations
- Development (publishedVersion)
- PubMed Central (publishedVersion)
- PubMed (publishedVersion)
- Funders
- National Institute of General Medical Sciences (T32GM007377)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Cell Biology › melanin and skin pigmentation
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Molecular Biology › Developmental Biology and Gene Regulation
- Life Sciences › Neuroscience › Developmental Neuroscience › Neurogenesis and neuroplasticity mechanisms
- Keywords
- FOXD3, MITF, Pax3, neural crest, melanoblasts, melanocyte differentiation, glial lineage, lineage switch, transcriptional repression, melanogenesis, neural crest migration, B16F10 melanoma
- MeSH terms
- Animals, Base Sequence, Cell Movement, Cells, Cultured, Chick Embryo, Embryo, Nonmammalian, Melanocytes, Molecular Sequence Data, Neural Crest, Neuroglia, Neurons, Promoter Regions, Genetic, Quail, Cell Lineage, Microphthalmia-Associated Transcription Factor, Paired Box Transcription Factors, Forkhead Transcription Factors, Cell Transdifferentiation
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