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Rationale: This case report presents the diagnosis and etiology of hilar/mediastinal lymphadenopathy in a male patient.Patient concerns: A 49-year-old man presented with fever and dyspnea after physical exertion. Diagnosis: The patient was diagnosed with melioidosis by cultivation of lymph node aspirate on blood agar using the VITEK 2 compact system. Interventions: The patient was treated with ceftazidime intravenously, combined with trimethoprim/sulfamethoxazole orally for 1 week. Once the patient was discharged, he began a 12-week course of trimethoprim/sulfamethoxazole. Outcomes: The patient recovered after treatment with ceftazidime and trimethoprim/sulfamethoxazole. Conclusions: Melioidosis is an infectious disease that mainly occurs in tropical regions. It can cause severe sepsis and pneumonia, and the infection in some patients may become chronic. Endobronchial ultrasound-transbronchial needle aspiration is a useful technique in the diagnosis of patients with hilar/mediastinal lymphadenopathy.