ArticleScientific reports2024
Loss of Pax3 causes reduction of melanocytes in the developing mouse cochlea.
Article in Scientific reports, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 7 citations in OpenAlex.
- Blue eyes in Felis catus: genetics and health impacts.Companion animal health and genetics · 2026Review
- Prevalence and Clinical Characteristics of Vitiligo-Associated Hearing Loss Among Patients in a Phase 2 Clinical Trial of Povorcitinib.Dermatology and therapy · 2026Article
- Insights Into Stria Vascular Function, Stria Immunity, and Age-Related Hearing Loss.Journal of the Association for Research in Otolaryngology : JARO · 2025Review
- Molecular differences between young and mature stria vascularis from organotypic explants and transcriptomics.iScience · 2025Article
- Expression of S100β during mouse cochlear development.European journal of histochemistry : EJH · 2025Article
- Exosomes Derived from Hypertrophic Scar Fibroblasts Suppress Melanogenesis in Normal Human Epidermal Melanocytes.International journal of molecular sciences · 2024Article
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Authors and funding
12 authors at 4 institutions in 2 countries.
Funding
Abstract
Cochlear melanocytes are intermediate cells in the stria vascularis that generate endocochlear potentials required for auditory function. Human PAX3 mutations cause Waardenburg syndrome and abnormalities of skin and retinal melanocytes, manifested as congenital hearing loss (~ 70%) and hypopigmentation of skin, hair and eyes. However, the underlying mechanism of hearing loss remains unclear. Cochlear melanocytes in the stria vascularis originated from Pax3-traced melanoblasts and Plp1-traced Schwann cell precursors, both of which derive from neural crest cells. Here, using a Pax3-Cre knock-in mouse that allows lineage tracing of Pax3-expressing cells and disruption of Pax3, we found that Pax3 deficiency causes foreshortened cochlea, malformed vestibular apparatus, and neural tube defects. Lineage tracing and in situ hybridization show that Pax3
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