The pterygoid hamulus (PH), a miniature yet significant structure of the sphenoid bone, has been linked to orofacial discomfort referred to as “pterygoid hamulus syndrome.” Pterygoid hamulus syndrome, is an uncommon etiology of orofacial pain with multiple differential diagnosis. In this article we present a case report of a 47 ‑year‑old female patient who reported to King Saud Medical City (KSMC) with chief complaint of pain on right side of the face for 2–3 years. She was diagnosed with “pterygoid hamulus syndrome” and surgically operated, the same being documented in this article. We have provided a literature appraisal regarding the incidence, diagnosis, and management of this condition. Given the distinct treatment approach required for this condition compared to other pain syndromes in the same area, clinicians should consider the possibility of a diagnosis of pterygoid hamular syndrome.