ALDH1A1

aldehyde dehydrogenase 1 family member A1

Tier: Cilia-Associated Genes

Ensembl:
ENSG00000165092
UniProt:
P00352
OMIM:
100640
Synonyms:
ALDH1, ALDC, ALDH-E1, PUMB1, RALDH1, RALDH

Cilia effects upon perturbation of ALDH1A1

Loss-of-function effect:
Shorter cilia
Overexpression effect:
Longer

Phenotypes

Human ciliopathy phenotype:
Downregulation of ALDH1A1 mRNA in ciliated cells from COPD and cystic fibrosis patients (Shinjyo 2025).

Subcellular localization

airway ciliated cells, cytosol

References: PMID:40408364

Functional category

  • Metabolism
  • Motile cilium & axoneme
  • Ciliary assembly/disassembly

Function

ALDH1A1 is selectively expressed in airway ciliated cells and detoxifies reactive aldehyde species (acrolein, 4-HNE, MDA) generated by PM2.5/DEP exposure and oxidative airway injury. Aldh1a1-/- mice show aberrant cilia regeneration after naphthalene or DEP exposure with reduced acetylated α-tubulin (TUBA, Lys40) levels, disorganized cilia, impaired mucociliary clearance, and increased susceptibility to S. pneumoniae pneumonia. Mechanism: Michael addition of α,β-unsaturated aldehydes to Lys40 of α-tubulin competes with the acetylation required for ciliary microtubule stability. ALDH1A1 mRNA is downregulated in ciliated cells from patients with COPD and cystic fibrosis. Pharmacological activation of ALDH1A1 by Alda-1 restores MCC after oxidative injury. (Shinjyo et al. 2025, J Clin Invest, PMID 40408364)