RATIONALE: Although paracoccidioidomycosis is one of the most prevalent endemic mycoses in Latin American countries, where at least 10 million people are infected, the prevalence of paracoccidioidomycosis in China remains unknown because no related case has been reported, and its diagnosis is extremely challenging for local clinicians because of the complexity of disease progression and lack of specific evidence. PATIENT CONCERNS: Here, we report the first case of PCM in a male patient with a long-term latent infection in China. DIAGNOSIS: The results of special staining, immunohistochemistry, lymph node biopsy pathology, and metagenomic second-generation sequencing indicated that the patient was infected with Paracoccidioides brasiliensis. INTERVENTIONS: In this case, the patient was administered voriconazole 200 mg twice daily. OUTCOMES: After continuous treatment for 6 months, the patient's symptoms improved significantly, and the medication was discontinued. The outpatient follow-up revealed no discomfort. LESSONS: This case is of great value for the early diagnosis, treatment, and prevention of the spread of this emerging infectious disease in China.