Though anomalies of the superior belly of the omohyoid have been described in medical literature, absence of superior belly of omohyoid is rarely reported. Herein, we report a rare case of unilateral absence of muscular part of superior belly of omohyoid. During laboratory dissections for medical undergraduate students, unusual morphology of the superior belly of the omohyoid muscle has been observed in formalin embalmed male cadaver of South Indian origin. The muscular part of the superior belly of the omohyoid was completely absent. The inferior belly originated normally from the upper border of scapula, and continued with a fibrous tendon which ran vertically lateral to sternohyoid muscle and finally attached to the lower border of the body of hyoid bone. The fibrous tendon was about 1 mm thick and received a nerve supply form the superior root of the ansa cervicalis. As omohyoid mucle is used to achieve the reconstruction of the laryngeal muscles and bowed vocal folds, the knowledge of the possible anomalies of the omohyoid muscle is important during neck surgeries.