Disseminated histoplasmosis in a patient with common variable immunodeficiency: A coincidence or the result of T cell defects?

普通变异型免疫缺陷患者发生播散性组织胞浆菌病:是巧合还是T细胞缺陷的结果?

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Abstract

BACKGROUND: In endemic regions, histoplasmosis is often seen in hosts with defective cell mediated immunity. We report a case of disseminated histoplasmosis in a patient with common variable immunodeficiency (CVID), a disorder mainly characterized by B cell defects. CASE: A 35 year old male with CVID developed fever, headache, dyspnea and pancytopenia within few weeks of swimming in the Tennessee River. After a non-revealing initial evaluation he was transferred to a tertiary facility for fever of unknown origin, where massive splenomegaly was noted. Clinical course was complicated by hypoxia from extensive bilateral lung infiltrates requiring non-invasive ventilation. Urine and serum Histoplasma antigens were positive. He was treated with liposomal amphotericin B followed by itraconazole after clinical improvement within 48 h and discharged home by day 6. Fungal blood cultures sent on day 1 grew Histoplasma capsulatum on day 19. After 5 months splenomegaly completely resolved and he successfully completed one year of treatment with itraconazole. CONCLUSIONS: Our case highlights the significance of T cell defects in CVID. More research focusing on T cell defects in CVID is required to understand the extent of vulnerability to such intracellular pathogens in CVID.

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