ATOH1

atonal bHLH transcription factor 1

Ensembl:
ENSG00000172238
UniProt:
Q92858
OMIM:
601461
Synonyms:
BHLHA14, HATH1, MATH-1, MATH1

Cilia effects upon perturbation of ATOH1

Cilia number / % ciliated:
Increased Cilia Number/Decreased cilia number

Ciliogenesis screen results (4 screens)

  • Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381
  • Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-2.51) PMID:41160700

Subcellular localization

cilia associated gene, nucleus

Functional category

  • Ciliary assembly/disassembly
  • Neurogenesis & migration
  • Signaling (Hedgehog, GPCRs, ion channels)
  • Axon guidance & growth
  • Transcription regulation

Function

Controls Primary Cilia Formation to Allow for SHH- Triggered Granule Neuron Progenitor Proliferation. Atoh1-knockdown GNPs were less likely to display a primary cilium compared to control (PMID: 30695697). Morphological examinations revealed that the numbers of stereocilia bundles of both IHCs and OHCs in the Atoh1-treated animals were significantly greater than those of the untreated cochleae (https://doi.org/10.1371/journal.pone.0046355).

Model organism evidence

Danio rerio (1 reference)

Otolith precursor particles, initially distributed throughout the otic vesicle lumen, become tethered to the tips of hair cell kinocilia (tether cilia) at the otic vesicle poles, forming two otoliths.

PMIDs: 22461562