DZANK1 (double zinc ribbon and ankyrin repeat domains 1) is a protein essential for intracellular vesicle transport in photoreceptor cells. Mechanistically, DZANK1 functions as a binding partner of NINL (Ninein-like protein) and associates with complementary subunits of the cytoplasmic dynein 1 motor complex, which is required for minus-end directed cargo transport 1. This NINL-DZANK1 protein module is critical for proper assembly and folding of the dynein motor complex. Loss of DZANK1 in zebrafish results in dysmorphic photoreceptor outer segments, accumulation of trans-Golgi-derived vesicles, mislocalization of Rhodopsin and Ush2a proteins, and severely impaired retrograde melanosome transport 1. DZANK1 is clinically relevant to ciliopathies, as photoreceptor outer segments are specialized primary cilia dependent on dynein-based intracellular transport for formation and function 1. A chr20 inversion event affecting DZANK1 exons has been identified in association with posterior polymorphous corneal dystrophy in a mouse model, though haploinsufficiency of DZANK1 alone does not cause disease 2. DZANK1 remains relatively understudied despite its importance in photoreceptor development and ciliary function.