FAM149B1

family with sequence similarity 149 member B1

Ensembl:
ENSG00000138286
UniProt:
Q96BN6
OMIM:
618413
Synonyms:
KIAA0974

Cilia effects upon perturbation of FAM149B1

Cilia number / % ciliated:
No effect
Loss-of-function effect:
Longer cilia

Ciliogenesis screen results (3 screens)

  • Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-5.35) PMID:26167766
  • Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680
  • Roosing et al. 2015 (siRNA) [siRNA]: No effect PMID:26595381

Phenotypes

Mouse ciliopathy phenotype:
male infertility
Human ciliopathy phenotype:
Joubert syndrome 36

Ciliopathy associations

  • Joubert Syndrome
  • Skeletal Ciliopathy

Subcellular localization

basal body, cilia

Functional category

  • Ciliary assembly/disassembly

Function

Required for normal ciliary biology and that its deficiency results in impaired ciliary structure and a range of ciliopathy phenotypes in humans along the spectrum of Joubert syndrome.

Model organism evidence

Mus musculus (1 reference)

BROMI/TBC1D32 together with CCRK/CDK20 and FAM149B1/JBTS36 contributes to intraflagellar transport turnaround involving ICK/CILK1.

PMIDs: 35609210

C. elegans (1 reference)

xbx-4, a homolog of the Joubert syndrome gene FAM149B1, acts via the CCRK and RCK kinase cascade to regulate cilia morphology.

PMIDs: 34731674