Rationale: Melioidosis is an emerging infectious disease in Brazil and caused by Burkholderia pseudomallei, with high morbidity and mortality rates. A total of 28 melioidosis cases were reported in Brazil until 2015. The majority of melioidosis cases were reported in the Northwest region of Brazil and such cases were not previously detected in the Midwest region of Brazil. Patient concerns: A 42-year-old man was admitted with a non-productive cough, dyspnea, myalgia, diffuse abdominal pain. Pulmonary auscultation revealed a vesicular murmur, snoring sounds, and the presence of basal crackling rales in the left hemithorax. The patient evolved with several respiratory failures and he was diagnosed as the first case of community-acquired pneumonia with sepsis caused by B pseudomallei in Mato Grosso do Sul, Midwest state of Brazil. Diagnosis: The cell isolates were subjected to 16S rRNA gene sequencing to confirm the bacterial species. Interventions: Administration of trimethoprim/sulfamethoxazole and meropenem stabilized the clinical condition of the patient. Subsequently upon discharge, the patient was also treated with trimethoprim/sulfametothoxazole for a year. Outcome:We reported the first case of community-acquired pneumonia with sepsis caused by B pseudomallei in Mato Grosso do Sul, Midwest state of Brazil and the patient survived. Lessons: The emergence of melioidosis in the Midwest region is being neglected and underestimated and melioidosis must be considered of the differential diagnosis in community infections.