Rhabdomyosarcoma is very rare and highly malignant tumor of striated muscle with rapid progression and high incidence of recurrence. Author has experienced a case of rhabdomyosarcoma of the botryoid type involving the right maxillary sinus in a 28 year old Korean male soldier, who admitted to Capital Military Hospital. The biopsy specimen taken from antrum via Caldwell-Luc's approach, shows microscopically rhabdomyosarcoma (Botryoid type). Tried to treat the patient with triple combined therapy (intra-arterial chemotherapy with 5 FU, irradiation with Co⁶⁰ and necrotomy), which is thought to be effectful. The patient was survival till 14 months later of medication, and reviewed literatures.