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Rhabdomyoma is a rare benign soft tissue tumor deriving from striated muscle. It can be classified generally into two subtypes according to the location: cardiac rhabdomyoma and extracardiac rhabdomyoma. Cardiac rhabdomyoma manifests as well-defined solitary or multifocal nodules in heart consisting myocardial-like tumor cells with rich cytoplasm, which is more common in kids and associated with tuberous sclerosis (1). Extracardiac rhabdomyoma can be divided into three groups according to morphological features (adult, fetal, and genital types). The adult and fetal types mainly occur in the head and neck region, while the genital type usually occurs in the genital tract. To the best of our knowledge, rhabdomyoma is rarely found in the respiratory system and the visceral pleura. Only one case of pulmonary rhabdomyoma has been reported in the English literature (2). It is difficult to make a correct pre-operative diagnosis. Herein, we report the first case of rhabdomyoma originating from visceral pleura.