<p>Ethiopia is one of the countries with high burden of pulmonary tuberculosis and the number of cases of extra pulmonary is increasing. However, diagnostic evidence to initiate anti-tuberculosis treatment among extra pulmonary tuberculosis (EPTB) cases is not well known. We presented here a case of isolated tuberculosis epididymitis with scrotal sinus after developing nonspecific testicular symptoms for more than 2 months and review the literature. Tuberculous epididymitis is a form of urogenital tuberculosis that is particularly rare and has the same clinical manifestation that mimics other more common infectious causes of epididymitis, leading to delay in diagnosis. So, one should maintain a high-index of suspicion to accurately diagnose tuberculous epididymitis in individuals presenting with suggestive signs and symptoms.</p>