Primary multi-drug resistant extrapulmonary tuberculosis is an uncommon form of the disease, but it seems that by increasing the number drug resistant tuberculosis around the world, the number of cases of primary multi-drug resistant tuberculosis with extrapulmonary presentation also is going to rise. In this report, we describe a 19-year old, HIV negative man with primary multi-drug resistant TB lymphadenitis, presented with cervical lymphadenopathy and sinus discharge at the site of involved lymph nodes. The Acid Fast Bacilli (AFB) smear of sputum was negative but the AFB smear of discharged fluid as well as the excisional biopsy of the lymph nodes confirmed the M. tuberculosis infection. The patient underwent the treatment with a combination of isoniazide, clofazimine, pyrazinamide, ofloxacin and amikacin with promising results. By increasing the number of drug resistant tuberculosis patients around the world, appropriate diagnosis and treatment of different presentations of the disease need a special attention.