MAP6D1

Cilia effects upon perturbation of MAP6D1

Cilia number / % ciliated:

No effect

Loss-of-function effect:

Shorter cilia

Ciliogenesis screen results (2 screens)

• Wheway et al. 2015 (siRNA) [siRNA]: Ciliogenesis Defect (z=-2.26) PMID:26167766
• Breslow et al. 2018 (CRISPR) [CRISPR]: No Significant Effect PMID:29459680

Subcellular localization

cilia, cilia associated gene, neurons

Functional category

• Ciliary assembly/disassembly
• Actin & cytoskeleton regulation
• Signaling (Hedgehog, GPCRs, ion channels)
• Transition zone

Function

In vertebrates the microtubule-associated proteins MAP6 and MAP6d1 stabilize cold-resistant (22355359). MAP6d1 assembles stable microtubule doublets by recruiting tubulin dimers onto the lattice of the A-tubule to initiate the nucleation of the B-tubule. In neurons, MAP6d1 localises to the proximal part of primary cilia via its Mn-motif, with its loss resulting in shortened cilia, a characteristic of ciliopathies. (40617876) Deletion of MAP6D1 in neurons leads to shortened primary cilia and defective Sonic Hedgehog signalling, a hallmark of ciliopathies. (40617876) MAP6d1 localizes to the proximal part of primary cilia via Mn-motif; loss results in shortened cilia. Assembles stable microtubule doublets by recruiting tubulin dimers onto A-tubule lattice to initiate B-tubule nucleation.