BACKGROUND: Pterygoid hamulus syndrome, a painful oral and facial syndrome, has been described in literature to be correlated with morphological changes in the length of the pterygoid hamulus of the sphenoid bone. CLINICAL PRESENTATION: The current case report describes the treatment for severe, continuous pain in the posterior right palate. Despite numerous conservative treatments given to the patient, no improvement was seen. Cone beam computed tomography (CBCT) measurements revealed an elongation as well as a significant medial deviation of the lower extremity of the medial pterygoid plate. The surgical resection was performed under local anesthesia. The pain subsided two days after the surgery, and there were no relapses in the weeks that followed. CONCLUSION: The medial deviation of the hamulus appeared to be important in the etiology of this painful syndrome. Additional research based on CBCT measurements will be required.