Histoplasmosis is a deep mycosis caused by Histoplasma capsulatum, which is endemic in many areas of the world but is relatively rare in China. Although the majority of cases present as a mild to moderate flu-like disease requiring only supportive therapy, approximately 1% of patients experience more serious pulmonary and extrapulmonary disease, which can be life-threatening if diagnosis is delayed or the treatment is not initiated rapidly. Definitive diagnosis is usually made by a combination of culture, detection of the organism in tissues, measurement of antibodies, and detection of antigen. We present the case of a 51-year-old patient who presented with histoplasmosis only, with several ulcerated lesions in the oral cavity and without HIV infection, who did not show any detectable signs and symptoms of systemic disease or extra-oral manifestations. Histopathological analysis indicated a chronic inflammatory process with granulomas with yeast-like organisms. Isolation of H. capsulatum and molecular identification provided the definitive diagnosis. Treatment with oral itraconazole led to remission of the oral lesions. This is the first Chinese case report of localized histoplasmosis with lesions restricted to the mouth in an HIV-negative patient.
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