WDR35 is a core component of the intraflagellar transport complex A (IFT-A), essential for retrograde ciliary transport and GPCR entry into cilia 1. It localizes to cilia and centrosomes and is required for ciliogenesis in both primary and motile cilia 12. WDR35 associates with the CCT chaperone complex and regulates subcellular localization of acetylated tubulin in primary cilia through interactions with TCP1 and RagA proteins 3. Loss of WDR35 impairs ciliary protein trafficking and triggers TGFβ-ECM-integrin signaling that drives polycystic liver disease progression 4. Biallelic WDR35 mutations cause autosomal recessive ciliopathies including cranioectodermal dysplasia (CED/Sensenbrenner syndrome) and short-rib polydactyly syndromes 156. CED patients present with craniofacial dysmorphisms, skeletal deformities, growth retardation, ectodermal abnormalities, and organ involvement including nephronophthisis and hepatic fibrosis 52. WDR35 mutations also cause airway mucociliary clearance defects through disrupted motile cilia formation 2. High WDR35 expression in lung adenocarcinoma correlates with improved chemotherapy sensitivity and better overall survival, suggesting potential prognostic value 7.