Background <p>Cysticercosis is a parasitic infection caused by Taenia solium larvae, with pigs as intermediate hosts and humans as definitive hosts. It rarely occurs in the maxillofacial region. This report describes a case of cysticercosis of right temporalis muscle.</p> Case presentation <p>A 35-year-old female presented with the right facial pain and reduced mouth opening for six years. Clinical examination revealed no pain during lateral or protrusive jaw movements, no joint clicking or crepitus and negative Mahan test. MRI of TMJ showed an 11x9x10 mm cyst within the right temporalis muscle in infratemporal fossa, accompanied by adjacent soft tissue edema. Her rural background, pig exposure, and MRI findings strongly pointed to a diagnosis of cysticercosis cyst. She was treated with Albendazole, Prednisolone and Diclofenac. The patient’s mouth opening was improved from 12 mm to 35 mm after six weeks. A follow-up MRI confirmed the complete resolution of the lesion, and the patient was free from the disease.</p> Conclusion <p>This case report emphasizes the importance of considering cysticercosis in the differential diagnosis of patients presenting with chronic trismus. Radiographic investigations should be done prior to perform any procedure to confirm clinical diagnosis.</p>