To describe a fatal case of histoplasmosis-associated HLH in a patient receiving adalimumab and the role of skin biopsy in identifying the infection.
Approach:
Case evaluation: A 41-year-old man receiving adalimumab presented with systemic illness, cytopenias, hepatosplenomegaly, and findings fulfilling at least six HLH-2004 criteria.
Diagnostic testing: Bone marrow fungal stains were negative. Newly recognized acneiform papules were biopsied; histopathology showed numerous invasive organisms, and subsequent molecular testing identified Histoplasma capsulatum.
Key Findings:
The patient had severe hyperferritinemia, cytopenias, hypofibrinogenemia, hypertriglyceridemia, and hemophagocytosis.
Urine Histoplasma antigen was positive, and molecular testing of the skin biopsy confirmed H. capsulatum.
The patient had been treated with adalimumab; the article notes TNF-α inhibition increases risk of severe or disseminated histoplasmosis.
The case report describes the illness as fatal.
Interpretation:
The authors report that biopsy of subtle, newly recognized skin lesions revealed numerous invasive organisms, and subsequent molecular testing identified Histoplasma capsulatum despite negative bone marrow fungal staining.
Limitations:
This is a single case report.
Final natural killer cell function and HLH genetic panel results were not available in the medical record.
The provided article context ends during the treatment course and does not include further clinical details.
Conclusion:
In this fatal case of histoplasmosis-associated HLH in a patient treated with adalimumab, biopsy of subtle acneiform papules revealed invasive organisms, and subsequent molecular testing identified Histoplasma capsulatum.
