WDR47 is a microtubule-associated protein that plays critical roles in neuronal development, ciliary function, and cellular transport. The protein is essential for neuronal polarization, where it interacts with CAMSAP family proteins to stabilize microtubule minus ends and facilitate proper neurite remodeling during early neuronal development 1. WDR47 protects CAMSAP2-decorated microtubule minus ends from katanin-mediated severing, ensuring proper stabilization of the neuronal microtubule network 2. In ciliary biology, WDR47 coordinates with JHY and SPEF1 to maintain central pair microtubule stability in motile cilia, contributing to proper ciliary waveform regulation 3. The protein also regulates autophagy-related transport by negatively regulating RUSC2-mediated kinesin-1-dependent distribution of ATG9A vesicles to cell periphery 4. Clinically, bi-allelic variants in WDR47 cause a complex neurodevelopmental syndrome characterized by corpus callosum dysgenesis, microcephaly, and enlarged ventricles, with severity correlating to the degree of functional loss 5. WDR47 dysfunction impairs mitochondrial and microtubule homeostasis, leading to callosal neuron death. Additionally, WDR47 variants have been associated with cardiovascular laterality defects and identified as a potential therapeutic target in Alzheimer's disease 67.