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Case Reports
. 2024 Apr 15:15:1371490.
doi: 10.3389/fimmu.2024.1371490. eCollection 2024.

Sulfasalazine-induced drug reaction with eosinophilia and systemic symptoms (DRESS) coinfected with COVID-19 complicated by hemophagocytic lymphohistiocytosis: a case report

Mengmeng Li  1 , Furong Li  1 , Yang Dai  2 , Yunou Zhang Zeng  2 , Xiaomei Chen  2
Affiliations
Case Reports

Sulfasalazine-induced drug reaction with eosinophilia and systemic symptoms (DRESS) coinfected with COVID-19 complicated by hemophagocytic lymphohistiocytosis: a case report

Mengmeng Li et al. Front Immunol. .

Abstract

Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS) is characterized by a widespread maculopapular rash, lymphadenopathy, fever, and multisystem involvement. Conversely, hemophagocytic lymphohistiocytosis (HLH) is an infrequent yet critical condition presenting with fever, hepatosplenomegaly, cytopenias, coagulation abnormalities, and elevated inflammatory markers. The overlapping clinical and laboratory features between DRESS and HLH poses a significant diagnostic challenge. Secondary HLH (sHLH) typically occurs in adults triggered by viral infections, malignancies, rheumatologic diseases, or immune deficiencies. Recently, COVID-19 has also been identified as one of the triggers for sHLH. Herein, we present a case of Sulfasalazine-induced DRESS coinfected with COVID-19 that subsequently progressed into HLH. Our patient exhibited common hepatorenal and splenic involvement along with rare cholecystitis and appendicitis. However, a significant improvement was observed upon the addition of etoposide and azathioprine. We hypothesize that excessive activation of the immune system and cytokine storm due to DRESS combined with COVID-19 infection led to more extensive systemic damage resulting in HLH development. This highlights the potential for severe consequences when DRESS coincides with HLH during a COVID-19 infection.

Keywords: CMV; COVID-19; drug reaction with eosinophilia and systemic symptoms; ebv; hemophagocytic lymphohistiocytosis.

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Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

Figure 1
Figure 1
(A–C) Diffuse erythema and scale over face and the trunk, on admission; (a–c) Post-treatment improvement showing only scattered erythema on the face and almost complete improvement over the trunk.
Figure 2
Figure 2
(A) The gallbladder wall was edematous, thickened, and rough with a slightly blurred surrounding fat space suggesting acute cholecystitis, (B) The appendix was slightly thickened, about 1.2 cm at its thickest, with a blurred surrounding fat space and multiple striated shadows, (C) PET-CT scans did not reveal any definitive signs of malignancy.
Figure 3
Figure 3
A bone marrow biopsy was negative for malignancy and did not show evidence of hemophagocytosis.
See this image and copyright information in PMC

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