Infliximab is an inhibitory of tumor necrosis factor-alpha which is used successfully for the treatment of inflammatory bowel disease and rheumatic disease. It has various side effects including injection-induced reactions, immunosuppression, demyelinating diseases, and cardiac effects. One of the most serious side effects is tuberculosis. In particular, the immunosuppressant drugs have a high risk of reactivating latent tuberculosis infection. Its activation probably may occur as an extra-pulmonary and, occasionally, may result in an unusual infection. Herein, we report a 30-year-old male case treated with infliximab and suffered from isolated testicular tuberculosis.