CKAP2L

cytoskeleton associated protein 2 like

Ensembl:
ENSG00000169607
UniProt:
Q8IYA6
OMIM:
616174
Synonyms:
FLJ40629, RADMIS

Cilia effects upon perturbation of CKAP2L

Loss-of-function effect:
Longer cilia

Ciliogenesis screen results (3 screens)

  • Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-5.16) PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Elliott et al. 2025 (CRISPRa) [CRISPRa]: Disassembly Trigger (casTLE Effect=-2.03) PMID:41160700

Phenotypes

Mouse ciliopathy phenotype:
Ckap2l knockout mice: reduced male fertility, decreased sperm count, impaired sperm motility, abnormally elongated sperm flagella. Cellular: elongated primary cilia, shortened mitotic spindles, cytokinesis failure with multinucleation.
Human ciliopathy phenotype:
Filippi syndrome (FLPIS, OMIM 272440): autosomal recessive craniodigital syndrome with pre- and postnatal growth retardation, microcephaly, distinctive facial appearance, syndactyly of fingers and toes, intellectual disability. CKAP2L localises to centrosome, mitotic spindle, and ciliary basal body; CKAP2L loss elongates primary cilia in human and mouse cells; Ckap2l KO mice show elongated sperm flagella (Hussain et al. 2014, PMID 25439729; Lyu et al. 2025, PMID 41370039).

Ciliopathy associations

  • Filippi Syndrome

Subcellular localization

centrosome, cilia

Functional category

  • Ciliary assembly/disassembly
  • Actin & cytoskeleton regulation

Function

The reciprocal ciliary localization patterns of CKAP2L and DHX56 highlighting region-specific variations in ciliary composition between the dorsal and ventral brain regions., CKAP2L has localized in cilia (PMID: 40654709). At the cellular level, CKAP2L is a bona fide microtubule-associated protein that localizes to microtubule-based organelles, including the centrosome, mitotic spindle, and ciliary basal body. Depletion of CKAP2L leads to shortened mitotic spindles and cytokinesis failure, resulting in multinucleation. Furthermore, we uncover a conserved function for CKAP2L as a negative regulator of primary cilium length; its loss markedly increases ciliary length in both human and mouse cells. (41370039)

Model organism evidence

Mus musculus (1 reference)

Interestingly, Ckap2l knockout mice show no overt developmental abnormalities, with the exception of reduced male fertility, evidenced by decreased sperm count, impaired motility, and abnormally elongated flagella.

PMIDs: 41370039