Idiopathic scoliosis (IS) refers to a 3D rotation of the spine that occurs in the absence of underlying vertebral anomalies or obvious physiological defects. Despite affecting approximately 4% of the population, the etiology and pathogenesis of IS remain poorly understood, largely due to genetic heterogeneity and historical lack of appropriate developmental models. Recently, zebrafish has emerged as a powerful system for studying IS, owing to well-developed genetic resources and a natural susceptibility to spinal curvatures. Here, we summarize the utility of zebrafish as a genetic and biological model of IS, examine current faithful mutant IS models, and focus on their recent advances towards understanding core mechanisms governing both normal spine morphogenesis and the pathogenesis of IS-like spinal deformities.