The transcription factor Sox10 is required for the specification, migration and survival of all nonectomesenchymal neural crest derivatives including melanophores. sox10-/- zebrafish lack expression of the transcription factor mitfa, which itself is required for melanophore development. We demonstrate that the zebrafish mitfa promoter has sox10 binding sites necessary for activity in vitro, consistent with studies using mammalian cell cultures that have shown that Sox10 directly regulates Mitf expression. In addition, we demonstrate that these sites are necessary for promoter activity in vivo. We show that reintroduction of mitfa expression in neural crest cells can rescue melanophore development in sox10-/- embryos. This rescue of melanophores in sox10-/- embryos is quantitatively indistinguishable from rescue in mitfa-/- embryos. These findings show that the essential function of sox10 in melanophore development is limited to transcriptional regulation of mitfa. We propose that the dominant melanophore phenotype in Waardenburg syndrome IV individuals with SOX10 mutations is likely to result from failure to activate MITF in the normal number of melanoblasts.
Transcriptional regulation of mitfa accounts for the sox10 requirement in zebrafish melanophore development Available to Purchase
These authors contributed equally to this work and are joint first authors
These authors contributed equally to this work and are joint first authors
These authors contributed equally to this work and are joint first authors
These authors contributed equally to this work and are joint first authors
Stone Elworthy, James A. Lister, Tom J. Carney, David W. Raible, Robert N. Kelsh; Transcriptional regulation of mitfa accounts for the sox10 requirement in zebrafish melanophore development. Development 15 June 2003; 130 (12): 2809–2818. doi: https://doi.org/10.1242/dev.00461
Download citation file:
Sign in
Client Account
Sign in via your institution
Sign in via ShibbolethAdvertisement
The Company of Biologists Workshops

For the last 15 years, our publisher, The Company of Biologists, has provided an apt environment to inspire biology and support biologists through our Workshops series. Read about the evolution of the Workshop series and revisit JEB's experience with hosting the first Global South Workshop.
Call for papers – Lifelong Development: the Maintenance, Regeneration and Plasticity of Tissues

Development invites you to submit your latest research to our upcoming special issue – Lifelong Development: the Maintenance, Regeneration and Plasticity of Tissues. This issue will be coordinated by Guest Editors Meritxell Huch (Max Planck Institute of Molecular Cell Biology and Genetics, Germany) and Mansi Srivastava (Harvard University and Museum of Comparative Zoology, USA), working alongside our team of academic Editors. Submit your articles by 30 May 2025.
Meet our 2025 Pathway to Independence (PI) fellows

We are delighted to announce our third cohort of PI fellows - researchers whom we will be supporting as they transition from postdoc to Principal Investigator. Read about the eight talented fellows chosen, whom we're excited to be working with as they navigate the job market.
In preprints
Did you know that Development publishes perspectives on recent preprints? These articles help our readers navigate the ever-growing preprint literature. Together with our preprint highlights service, preLights, these perspectives help our readers navigate the ever-growing preprint literature. We welcome proposals for ‘In preprints’ articles, so please do get in touch if you’d like to contribute.
the Node: Have your say

Our community site, the Node, is conducting a user survey about the content and the design of the site. Help us shape the Node's future and thank you for being a part of the Node over the last 15 years.