The genitourinary tract is the most common site for extrapulmonary tuberculosis (TB). Penile TB is extremely rare comprising less than 1 % of all genital TB cases in males. It most commonly presents either as a superficial ulcer on the glans or around the corona. Diagnosis of penile TB is often difficult because it can mimic numerous other diseases. The association of TB with AIDS, and the increasing incidence of multiple drug resistance has further compounded the problem. The case described herein involves a patient with multidrug-resistant smear-positive penile TB that was undiagnosed initially due to the lack of clinical suspicion of TB, and once diagnosed failed to respond to first line antitubercular drugs because of multiple drug resistance.