Abstract: Objective: To report a case of histoplasma capsulatum with granulomatous hyperplasia occurring in labial-buccal and gingiva mucosa. Diagnosis and Treatment: This case involves a middle-aged male patient presenting with granulomatous hyperplasia of the gingiva and labial-buccal mucosa. The diagnosis of Histoplasma capsulatum infection was confirmed by pathological examination, fungal culture, and assisted by pathogenic microorganism nucleic acid high-throughput sequencing. After excluding fungal infections of the lungs and other organs, a treatment regimen was formulated including oral itraconazole capsules for 6 weeks combined with local antifungal therapy. Results: Five weeks after treatment, the granulomatous ulcers of the patient’s gingiva and lips healed completely. Fungal culture results were negative, which met the criteria for drug withdrawal, with no recurrence during follow-up. Conclusions: For oral granulomatous ulcers that are persistent and unresponsive to conventional treatment, especially in patients with a history of fungal exposure or local immune disorders, the possibility of orofacial Histoplasma capsulatum infection should be considered.

Key words: Histoplasma capsulatum infection, granulomatous ulcer, fungal infection, fungal culture, itraconazole