Temporalis Muscle Hypertrophy (TMH) is a rare entity of masticatory muscle hypertrophy. It is a disease of important differential diagnosis between peripheral nervous system dysfunctions and neuromuscular diseases. TMH is most commonly bilateral and usually associated with other types of masticatory muscles hypertrophy, such as masseter or pterygoid hypertrophy. However, isolated unilateral TMH is extremely rare. After collecting data from the electronic medical record and allowing the patient permission to report the anamnesis, we describe the clinical case of an unusual form of pain and facial edema. In this case report, we present an adult patient with unilateral temporalis muscle hypertrophy.
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