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

Subcellular localization

No ciliary localization reported (cilia-associated gene).

References: PMID:31723061

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 anatomical pathologies, with concomitant rescue of the overabundance of key signaling molecules ( 31723061).