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Sarcocystis is an apicomplexan protozoan, such as Toxoplasma and Plasmodium. Human muscular sarcocystis infection is a rarely reported case. The rarity of the reported cases can be attributed to nonspecific symptoms and nonrecognition of the protozoa on histology. Sarcocystis infection has been catching up recognition in Southeast Asian countries. It is a food-borne zoonosis. There are more than hundred Sarcocystis spp. known and most have been isolated from muscle tissues of various intermediate hosts, including mammals, birds, and reptiles. They are parasites with dual hosts to accommodate their dual life cycles. Humans are the accidental intermediate hosts, as have been reported as intramuscular sarcocysts of unknown species. We report a similar case of 27-year-old male, who underwent wide local excision of Ewing's sarcoma of the right arm, in conjunction with which muscular sarcocystis infection was identified. We report a rare case of muscular sarcocystis in a rarer combination with Ewing's sarcoma, with no such previous case in the literature available.