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<i>Mycobacterium chimaera</i> is a slow-growing, nontuberculous mycobacterium (NTM) belonging to the <i>Mycobacterium avium complex</i> (<i>MAC</i>). It was identified as a unique species in 2004. Since 2013 it has been reported as a cause of disseminated infection in patients after cardiac surgeries. Only a few cases associated with underlying lung diseases have been noted. <i>M. chimaera</i> infection is characterized by ambiguous symptoms. There is no treatment with proven effectiveness, and it has a poor prognosis. Silicosis is a disease that can predispose to mycobacterial infection. Silica damages pulmonary macrophages, inhibiting their ability to kill mycobacteria. We present a case of <i>M. chimaera</i> infection in a patient with silicosis and without other comorbidities. To our knowledge, it is the first case of silicosis associated with <i>M. chimaera</i> disease. A 45-year-old man presented with a persistent low-grade fever. Based on the clinical and radiological picture, positive cultures, and histological examination, the nontuberculous mycobacterial disease was diagnosed. First, multidrug therapy according to the treatment guidelines for <i>MAC</i> was implemented, then antibiotics were administrated, based on drug sensitivity. Despite the treatment, eradication was not achieved and the patient died. The analysis of <i>M. chimaera</i> infection cases could contribute to developing recommendations and thus improve the prognosis.