Background: Omohyoid muscle syndrome (OMS) (not omohyoid syndrome) is a rare clinical condition that has a characteristic feature of a protruding lateral neck mass during swallowing. The use of endoscopic surgery on the neck is now pretty well established for thyroid and parathyroid glands. Patients with OMS usually undergo simple surgical transection of the omohyoid muscle. The procedure leaves operative scars on the neck, and most patients worry about the cosmetic problems. We report here the first use of an endoscopic procedure instead of traditional surgery for treatment of OMS. Materials and Methods: We present a rare case of a 26-year-old Chinese man who noted a protruding mass involving the right side of his neck during the past 10 years. OMS was diagnosed. Laparoscopic simple transection of the omohyoid muscle by an ultrasonically activated scalpel was performed. Results: After laparoscopic transection of the omohyoid muscle, the neck mass completely disappeared during swallowing, and there were no operative scars on the neck. Conclusions: To our knowledge, this is the first report of laparoscopy for treatment of OMS. We believe that the laparoscopic procedure is made acceptable for this unusual disease because of the cosmetic result.