Benign hypertrophy of the masseter muscle is a relatively uncommon condition which may occur unilaterally or bilaterally. The etiology of the ailment is unclear in the majority of cases, and the histopathological features are usually those of normal muscle. A case of unilateral masseter muscle enlargement is described in a 19-year-old Arab male. The histopathological findings were compatible with a mild myopathic disorder. The characteristics, differential diagnosis, etiology, treatment and histopathological features of masserteric hypertrophy are discussed.