USP38

Cilia effects upon perturbation of USP38

Ciliogenesis screen results (5 screens)

• Kim2016: No effect
• Wheway et al. 2015 (siRNA) [siRNA]: No effect PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: Positive Regulator (Hh signaling, casTLE effect=-0.75) PMID:29459680
• Roosing et al. 2015 (siRNA) [siRNA]: Hyper-ciliogenesis (robust z=3.02, ciliated=60.8%) PMID:26595381
• Kim et al. 2010 (siRNA) [siRNA]: Longer Cilia (Area per Cilia z=5.03) PMID:20393562

Phenotypes

Mouse phenotype:

decreased exploration in new environment, increased total body fat amount, decreased lean body mass, decreased circulating creatinine level, decreased prepulse inhibition, abnormal bone structure, thrombocytopenia, prolonged rr interval, increased mean corpuscular volume

Mouse ciliopathy phenotype:

persistence of hyaloid vascular system

Subcellular localization

cilia associated gene

Functional category

• Ciliary assembly/disassembly
• Trafficking (BBSome, small GTPases, vesicular transport, ATPases)

Function

USP35 (and its orthologue USP38) showed that suppression or ablation in vivo rescued a host of ciliopathy-associated a tomical pathologies, with concomitant rescue of the overabundance of key sig ling molecules ( 31723061).