Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient

Background: The search for the trigger of hemophagocytic lymphohistiocytosis (HLH) and its management can be challenging in immunocompromised patients. In HLH triggered by infection, immunosuppressive therapy is desired to suppress hyperinflammation, but may worsen the underlying infection. Objectiv...

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Main Authors: N.El Bardai, L.M. Vos, H.L. Leavis, M. Jak, A.H.W. Bruns, M. Limper
Format: Article
Language:English
Published: Elsevier 2025-04-01
Series:Clinical Infection in Practice
Online Access:http://www.sciencedirect.com/science/article/pii/S2590170225000810
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author N.El Bardai
L.M. Vos
H.L. Leavis
M. Jak
A.H.W. Bruns
M. Limper
author_facet N.El Bardai
L.M. Vos
H.L. Leavis
M. Jak
A.H.W. Bruns
M. Limper
author_sort N.El Bardai
collection DOAJ
description Background: The search for the trigger of hemophagocytic lymphohistiocytosis (HLH) and its management can be challenging in immunocompromised patients. In HLH triggered by infection, immunosuppressive therapy is desired to suppress hyperinflammation, but may worsen the underlying infection. Objectives: To review available literature on the management and prognosis of immunocompromised patients with disseminated histoplasmosis induced HLH. Methods: We describe a case and review all previously reported cases of disseminated histoplasmosis induced HLH on PubMed until June 2023. Results: The literature review yielded 30 cases. Antifungal therapy was administered in all except one patient. In 18 cases, HLH was treated with immunosuppressive therapy. The most common immunosuppressive regimens were corticosteroid monotherapy and corticosteroids combined with etoposide. This case report is the first to describe the use of anakinra. The overall mortality rate was 23.3 % (n = 7). Conclusions: Disseminated histoplasmosis induced HLH is a severe disorder, requiring prompt recognition and immediate intervention. Currently, no specific treatment protocol exists for this disorder. Treatment should be tailored to the patient’s condition, symptoms and individual characteristics. Careful consideration regarding immunosuppressive therapy for HLH is needed to prevent worsening of the infection. Additionally, anticipating the progression of the disease is necessary to ensure timely and appropriate interventions.
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series Clinical Infection in Practice
spelling doaj-art-57a0ff8c969141eaa208c2ef07bc78092025-08-20T03:21:55ZengElsevierClinical Infection in Practice2590-17022025-04-012610048810.1016/j.clinpr.2025.100488Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patientN.El Bardai0L.M. Vos1H.L. Leavis2M. Jak3A.H.W. Bruns4M. Limper5University Medical Centre Utrecht, Department of Rheumatology and Clinical Immunology, Utrecht, Netherlands; Corresponding author at: Heidelberglaan 100, 3584 CX Utrecht, Netherlands.University Medical Centre Utrecht, Department of Internal Medicine and Infectiology, Utrecht, NetherlandsUniversity Medical Centre Utrecht, Department of Rheumatology and Clinical Immunology, Utrecht, NetherlandsUniversity Medical Centre Utrecht, Department of Internal Medicine and Haematology, Utrecht, NetherlandsUniversity Medical Centre Utrecht, Department of Internal Medicine and Infectiology, Utrecht, NetherlandsUniversity Medical Centre Utrecht, Department of Rheumatology and Clinical Immunology, Utrecht, NetherlandsBackground: The search for the trigger of hemophagocytic lymphohistiocytosis (HLH) and its management can be challenging in immunocompromised patients. In HLH triggered by infection, immunosuppressive therapy is desired to suppress hyperinflammation, but may worsen the underlying infection. Objectives: To review available literature on the management and prognosis of immunocompromised patients with disseminated histoplasmosis induced HLH. Methods: We describe a case and review all previously reported cases of disseminated histoplasmosis induced HLH on PubMed until June 2023. Results: The literature review yielded 30 cases. Antifungal therapy was administered in all except one patient. In 18 cases, HLH was treated with immunosuppressive therapy. The most common immunosuppressive regimens were corticosteroid monotherapy and corticosteroids combined with etoposide. This case report is the first to describe the use of anakinra. The overall mortality rate was 23.3 % (n = 7). Conclusions: Disseminated histoplasmosis induced HLH is a severe disorder, requiring prompt recognition and immediate intervention. Currently, no specific treatment protocol exists for this disorder. Treatment should be tailored to the patient’s condition, symptoms and individual characteristics. Careful consideration regarding immunosuppressive therapy for HLH is needed to prevent worsening of the infection. Additionally, anticipating the progression of the disease is necessary to ensure timely and appropriate interventions.http://www.sciencedirect.com/science/article/pii/S2590170225000810
spellingShingle N.El Bardai
L.M. Vos
H.L. Leavis
M. Jak
A.H.W. Bruns
M. Limper
Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
Clinical Infection in Practice
title Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
title_full Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
title_fullStr Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
title_full_unstemmed Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
title_short Disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
title_sort disseminated histoplasmosis induced hemophagocytic lymphohistiocytosis in an immunocompromised patient
url http://www.sciencedirect.com/science/article/pii/S2590170225000810
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