Multiple recurrences of Talaromyces marneffei infection with HLH in an HIV-negative patient: a case report

BACKGROUND: Talaromycosis complicated by hemophagocytic lymphohistiocytosis (HLH) is a life-threatening condition associated with high mortality, yet the management of recurrent disease in HIV-negative patients is not well defined. CASE PRESENTATION: We report a 49-year-old man who presented with fever, lymphadenopathy, and an uncommon ulcerative scalp lesion that has been poorly documented in HIV-negative talaromycosis. Talaromyces marneffei (T. marneffei) infection was confirmed by blood culture, and HLH was subsequently diagnosed. Initial induction therapy with amphotericin B deoxycholate (AmB-D) followed by itraconazole (ITZ) maintenance resulted in clinical improvement; however, the patient experienced multiple recurrences over three years, including a recurrence while on ITZ. Immunological evaluation later revealed positive anti-interferon-γ autoantibodies (AIGA). Re-induction with liposomal amphotericin B (L-AmB) followed by maintenance posaconazole (PCZ) resulted in short-term remission, with no recurrence during three months of follow-up. CONCLUSIONS: This case demonstrates the successful management of recurrent talaromycosis with L-AmB induction followed by PCZ maintenance in an HIV-negative patient with AIGA-associated immunodeficiency presenting with initial HLH, highlighting the importance of early diagnosis, individualized antifungal strategies, and the potential utility of PCZ as maintenance therapy in high-risk patients.