Purpose: To report a rare case of angioleiomyoma of the ciliary body Methods :The clinical manifestation, imaging findings, histopathologic characteristics were analyzed in a 32-year-old male patient with angioleiomyoma of the ciliary body. Results:The tumor was removed intact with local resection. Histopathologic examination revealed that the tumor was full of vessels and it was composed of spindle cells with abundant cytoplasm. Immunohistochemical studies showed positive for SMA and Desmin and negative for S100 and HMB-45. Conclusions: Angioleiomyoma of the ciliary body is a rare tumor that can be successfully treated with local surgical resection in this area. It needs to be differentiated from other tumors, especially malignant melanoma. Eye Science 2004;20:19-22.