Myiasis which is a parasitic disease of humans and vertebrates, is caused by dipterous fly larvae feeding on the host's necrotic or living tissue. Although infestation by fly larvae is much more prevalent in animals, it occurs relatively frequently in humans in rural, tropical and subtropical regions of Africa and America. Myiasis is usually associated with poor general health and hygiene, restricted mobility and ulcerating lesions. The pathophysiology of the human infection differs depending on the fly species and where the larvae are located. It could be external or internal, and the invasion by the maggot could be obligatory, facultative and sometimes acci-dental. Myiasis is a self-limiting infestation with minimal morbidity in the vast majority of cases. Urogenital myiasis, associated with urinary obstruction, poor hygiene of the local site and ulcerating lesions has been infrequently reported. In this report, a case of 29 year-old male patient who presented with genitourinary myiasis caused by Psychodo albipennis (Diptera: Nematocera), was presented. The patient was admitted to the hospital with the complaints of urinary incontinence of one week duration and presence of small, thin, motile, grayish-white objects in his urine. Physical examination, blood and urine examination and stool microscopy revealed no pathology. No growth was detected in his urine culture. The examination of discharged larva in urine sample at Refik Saydam National Public Health Agency, Parasitology Laboratory led to the diagnosis of urogenital myiasis. No risk factor was identified in the patient who had proper hygienic conditions, was living in urban area and was of high socioeconomic status. This case was presented to withdraw attention to myiasis which is frequent in Turkey, however, is usually overlooked.