We present a case of a 34-year-old male patient who met the criteria for 11.8 Headache or facial pain attributed to inflammation of the stylohyoid ligament (Headache Classification Committee of the International Headache Society, 2018) and underwent surgical treatment, resulting in complete relief of symptoms. We discuss how these patients may present clinically, how to diagnose them, and the therapeutic possibilities. This case report emphasizes the importance of diagnosing this rare condition, its complications, and raising awareness among healthcare professionals to provide relief to patients who may have their lives significantly impaired by the presented symptomatology.