International Journal of Case Reports and Images (IJCRI) is an international, peer reviewed, monthly, open access, online journal, publishing high-quality, articles in all areas of basic medical sciences and clinical specialties.Aim of IJCRI is to encourage the publication of new information by providing a platform for reporting of unique, unusual and rare cases which enhance understanding of disease process, its diagnosis, management and clinico-pathologic correlations. Primary leiomyosarcoma of the testis is a rare entity with only less than 20 cases were reported. The standard therapy is difficult to recommend because of the rarity of this tumor. Case Report: We reported a case of primary intratesticular leiomyosarcoma in a 78-yearold male with unremarkable past medical history. The patient was presented within isolated testicular enlargement. Ultrasound revealed a large solid, homogenous and hyper echoic mass of the right testicle. Lactate dehydrogenase (LDH) was elevated, however, serum alphafetoprotein (AFP) and beta-human chorionic gonadotropin (β-hCG) were within normal limits. A right radical orchidectomy was performed. The diagnosis of primary leiomyosarcoma of testis was confirmed by histopathology.
Conclusion:The primary leiomyosarcoma of testis is a rare diagnosis which should be one of the differential diagnoses of testicular mass with normal tumor markers.