Mycobacterium kansasii extrapulmonary infections are infrequent in immunocompetent adults. Rifampin (RIF), clarithromycin (CLR), isoniazid (INH) and ethambutol (EMB) are included in all the standard regimens against M.kansasii. We report a case of a healthy 65-year-old male farmer who presented with isolated right supraclavicular lymphadenopathy. The lymph node FNA showed acid-fast-bacilli and granulomatous inflammation. Quantiferon TB Gold test, HIV serology, and functional immunological studies were all negative or normal. He was put on a standard 4 drugs anti-tuberculous regimen that was switched to RIF + CLR+ INH after the Microbiology lab demonstrated an EMB-resistant Mycobacterium kansasii isotype I strain. The patient was cured after 12 months of therapy. This is the 6th reported case of M. kansasii extrapulmonary lymphadenitis in an immunocompetent adult and the 2nd showing EMB resistance in the world literature. Antimycobacterial regimens against M. kansasii, classically resistant to pyrazinamide (PZA) might also exclude EMB due to its increasing resistance in Europe. A 612 months therapy with at least 2 effective antimycobacterial drugs including RIF + CLR might be enough to treat extrapulmonary M. kansasii infections in immunocompetents.