A 42-year-old man with unilateral agenesis of seminal vesicle associated with contralateral hypoplasia of seminal vesicle is presented. The patient was referred for evaluation of infertility. Physical examination was normal. Semen analysis yielded azoospermia with low seminal volume of 1 mL. Transrectal ultrasonography (TRUS), Computed Tomography (CT) and Magnetic Resonance Imaging (MRI) demonstrated absence of left seminal vesicle and hypoplastic right seminal vesicle. TRUS may be a good method for initial evaluation of these such anomalies. However, CT and MRI should be reserved for the cases whose TRUS results are not conclusive. Assisted reproductive techniques were recommended to the patient for children.