Hematuria is defined as the abnormal presence of red blood cells in the urine and is commonly divided into gross and microscopic hematuria. Terminal hematuria occurs at the end of the urine stream and may have a prostatic, bladder, or trigonal cause. Here, we present a 16-year-old boy who developed terminal hematuria after 1 month of treatment with isotretinoin for acne vulgaris. This side effect of isotretinoin is not reported in the literature. This is the first case report.