Ultrastructural pathological changes in the cochlear cells of connexin 26 conditional knockout mice
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Updated: May 9, 2026

Evaluation of Planar-Cell-Polarity Phenotypes in Ciliopathy Mouse Mutant Cochlea
Published on: February 21, 2016
Lan Lin1, Yun-Feng Wang, Shu-Yi Wang
1Department of Pathology, Eye and ENT Hospital of Fudan University, Shanghai 200031, P.R. China.
Abstract:
Mutations in the gene of connexin 26 (Cx26) are the most common cause of human non‑syndromic hereditary deafness. The pathogenesis of deafness caused by Cx26 remains uncertain. To explore the basic mechanism underlying Cx26 null mutations, ultrastructural changes and a number of marker proteins in the cochlear sensory epithelium of Cx26 conditional knockout mice were observed in the current study. Cochlear specimens were obtained from Cx26 conditional knockout mice (cCx26ko), while wild‑type mice served as controls. Antibodies against the pillar cell marker P75, the supporting cell marker prox1 and hair cell markers myosin 6 and phalloidin were labeled in different cells of the cochlear sensory epithelium of cochlear cryosections. The ultrastructural morphology of cochlear sensory epithelium was observed using transmission electron microscopy. Following the observation of cochlear sensory epithelium cell markers for hair cells and supporting cells, no significant changes were observed at the early stage, while the tunnel of the organ of Corti and Nuel's space was not developed prior to hearing onset in cCx26 knockout mice. Cell death was observed from postnatal day 10 (P10). The only region of surviving cells observed in the cochlea was the Hensen cell region, where microglia‑like cells appeared following P180. Overall, the present study showed an abnormal ultrastructural morphology in the cochlear sensory epithelium in cCx26ko mice. Microglia‑like cells may be involved in the process of cell degeneration in cCx26ko mice.
Insights
Connexin 26 (Cx26) gene mutations cause hereditary deafness. Cx26 knockout mice show abnormal cochlear ultrastructure and cell death, suggesting microglia-like cells may contribute to degeneration.
Area of Science:
- Genetics
- Otolaryngology
- Cell Biology
Background:
- Mutations in the connexin 26 (Cx26) gene are the primary cause of non-syndromic hereditary hearing loss.
- The precise mechanisms driving deafness associated with Cx26 mutations are not fully understood.
Purpose of the Study:
- To investigate the ultrastructural changes and cellular markers in the cochlear sensory epithelium of Cx26 conditional knockout mice.
- To elucidate the underlying mechanisms of deafness resulting from Cx26 null mutations.
Main Methods:
- Examined cochlear specimens from Cx26 conditional knockout (cCx26ko) mice and wild-type controls.
- Utilized transmission electron microscopy for ultrastructural analysis.
- Employed antibodies for pillar cell (P75), supporting cell (prox1), and hair cell (myosin 6, phalloidin) markers.
Main Results:
- Early stages showed no significant changes in hair and supporting cell markers.
- The tunnel of the organ of Corti and Nuel's space failed to develop before hearing onset in cCx26ko mice.
- Cell death initiated around postnatal day 10 (P10), with microglia-like cells appearing in surviving Hensen cells post-P180.
Conclusions:
- Cx26 deficiency leads to abnormal ultrastructural morphology in the cochlear sensory epithelium.
- Microglia-like cells may play a role in the degenerative processes observed in Cx26-deficient cochleas.

