MYO1D

Cilia effects upon perturbation of MYO1D

Cilia number / % ciliated:

Decreased cilia number

Loss-of-function effect:

Shorter cilia

Ciliogenesis screen results (3 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-2.78) 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 phenotype:

decreased startle reflex, decreased bone mineral density

Mouse ciliopathy phenotype:

abnormal auditory brainstem response

Subcellular localization

cilia associated gene

Functional category

• Ciliary assembly/disassembly
• Actin & cytoskeleton regulation

Function

Myo1D controls the orientation of LRO cilia and interacts functio lly with the pla r cell polarity (PCP) pathway component VanGogh-like2 (Vangl2), to shape a productive LRO flow. Our findings identify Myo1D as an evolutio rily conserved regulator of animal LR asymmetry, and show that functio l interactions between Myo1D and PCP are central to the establishment of animal LR asymmetry ( 29769531).