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Oculo-auriculo-vertebral spectrum (OAVS) is a complex disorder that is represented by wide variations in phenotypic presentation. Recent publications and systematic reviews of the available literature are presented here.Treatment strategies vary among craniofacial centers across the country. Advances in presurgical planning, virtual surgical planning, and computer-aided manufacturing have been incorporated in the treatment of patients. The psychosocial effect of OAVS with longitudinal follow-up is now being studied.Optimal evaluation and management of the OAVS patient requires an awareness of the phenotypic and genetic differences and involves a multidisciplinary team in order to effectively and appropriately diagnose and treat such patients.