Historically, Mycobacterium bovis infection in humans was associated with consumption of unpasteurized milk and dairy products and this is still the most important route of exposure in limited-resource countries; in rich resource countries, this infection has been usually observed in elder subjects. Direct airborne transmission from animals to humans is thought to be a rare as well as an interhuman transmission. We here describe the diagnosis and clinical management of pulmonary M. bovis infection in a 30-year-old healthy Italian female patient. One interesting aspect of this case is the absence of identifiable risk factors including the consumption of unpasteurized diary products or animal contact. Being M. bovis usually resistant to pyrazinamide, it is important to understand the epidemiology of human Mycobacterium bovis tuberculosis in order to personalize anti tubercular treatment.