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Case Reports
. 2022 Dec 28:2022:9521128.
doi: 10.1155/2022/9521128. eCollection 2022.

Two Cases of Hemophagocytic Lymphohistiocytosis Associated with Disseminated Histoplasmosis Presented with Transient Pancytopenia

Novi Apriany  1 , Usi Sukorini  2 , Tri Ratnaningsih  2 , Rizka Humardewayanti Asdie  3 , Yanri Wijayanti Subronto  3 , Susanna Hilda Hutajulu  4 , Ibnu Purwanto  4 , Mardiah Suci Hardianti  4
Affiliations
Case Reports

Two Cases of Hemophagocytic Lymphohistiocytosis Associated with Disseminated Histoplasmosis Presented with Transient Pancytopenia

Novi Apriany et al. Case Rep Med. .

Abstract

Transient pancytopenia due to reactive bone marrow suppression often occurs in hemophagocytic lymphohistiocytosis (HLH), a syndrome resulting from excessive immune activation following a severe infection. We reported two cases with pancytopenia and disseminated histoplasmosis accompanied by HLH, initially suspected to be blood malignancies. Our first case documented the relevance between the improvement of pancytopenia and the clearance of Histoplasma capsulatum in serial bone marrow aspirations. The second case showed immense Histoplasma engulfment by the macrophage in relation to a severe clinical condition, followed by improvement of clinical symptoms in accordance with the recovery of pancytopenia. These two cases highlighted the importance of comprehensive and critical analysis for cases with concurrent pancytopenia and severe infection, since it may be that the pancytopenia underlies the severe infection or vice versa.

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Conflict of interest statement

The authors declare that there are no conflicts of interest.

Figures

Figure 1
Figure 1
Initial bone marrow aspiration showing myelodysplasia syndrome (MDS) with refractory cytopenia with multilineage dysplasia (RCMD) subtype and infection with H. capsulatum inside the macrophages (taken 2 weeks after admission and as the initial point to start antifungal treatment).
Figure 2
Figure 2
Bone marrow aspiration after 4 weeks of antifungal treatment showed the reduction of intracellular budding yeast of Histoplasma inside the macrophages.
Figure 3
Figure 3
Bone marrow aspiration after 10 weeks of antifungal treatment showed macrophages without Histoplasma.
Figure 4
Figure 4
Improvement in peripheral blood parameters following treatment with an antifungal in case 1.
Figure 5
Figure 5
Large ecchymosis in the lower extremities.
Figure 6
Figure 6
Bone marrow aspiration at the time of diagnosis showed Histoplasma capsulatum yeasts inside the macrophages.
Figure 7
Figure 7
Histoplasma capsulatum yeast in methylene blue (a) and KOH (b) staining taken 5 weeks after starting BMA culture in Sabouraud agar.
Figure 8
Figure 8
Improvement in peripheral blood parameters following treatment with an antifungal in case 2.
See this image and copyright information in PMC

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