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Hemophagocytic lymphohistiocytosis in a patient with COVID-19 treated with tocilizumab: a case report.
J Med Case Rep. 2020 Oct 15; 14(1):187.JM

Abstract

BACKGROUND

The understanding of coronavirus disease 2019 (COVID-19) is rapidly evolving. Although it is primarily a respiratory illness, other manifestations, such as Guillain-Barré syndrome, immune thrombocytopenia, and immune-mediated thrombotic thrombocytopenic purpura, have been described. We present a case of a patient with hemophagocytic lymphohistiocytosis secondary to COVID-19 treated with tocilizumab with a marked biochemical improvement.

CASE PRESENTATION

In this case report we present a Caucasian patient with COVID-19 who developed a marked elevation of inflammatory parameters with ferritin 36,023 μg/L, but also elevated C-reactive protein 334 mg/L and lactate dehydrogenase 1074 U/L, 1 week after admission to the intensive care unit. He met five of eight criteria for hemophagocytic lymphohistiocytosis, but he lacked the high fever and cytopenia seen in the majority of cases. He was treated with tocilizumab, a monoclonal antibody targeting the interleukin-6 receptor, and over the next days, a rapid decrease in ferritin and C-reactive protein levels was observed. However, his respiratory failure only improved gradually, and he was weaned off the respirator 11 days later.

CONCLUSION

COVID-19 may induce a hyperinflammatory clinical picture and in some cases develop into hemophagocytic lymphohistiocytosis. In our patient's case, therapeutic interleukin-6 blockade abrogated signs of hyperinflammation but did not seem to improve pulmonary function. Measurement of ferritin and C-reactive protein, as well as quantification of interleukin-6 on indication, should be performed in patients with severe COVID-19. Specific treatment in such patients must also be contemplated, preferably in randomized controlled trials.

Authors+Show Affiliations

Department of Internal Medicine, Molde Hospital, Molde, Norway. birgitte.tholin@helse-mr.no.Department of Internal Medicine, Molde Hospital, Molde, Norway. Department of Microbiology, Molde Hospital, Molde, Norway.Research Institute of Internal Medicine, Oslo University Hospital and University of Oslo, Oslo, Norway. Section of Clinical Immunology and Infectious Diseases, Oslo University Hospital, Oslo, Norway.Department of Anesthesiology, Molde Hospital, Molde, Norway.Section of Hematology, Department of Internal Medicine, Haukeland University Hospital, Bergen, Norway.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

33054818

Citation

Tholin, Birgitte, et al. "Hemophagocytic Lymphohistiocytosis in a Patient With COVID-19 Treated With Tocilizumab: a Case Report." Journal of Medical Case Reports, vol. 14, no. 1, 2020, p. 187.
Tholin B, Hauge MT, Aukrust P, et al. Hemophagocytic lymphohistiocytosis in a patient with COVID-19 treated with tocilizumab: a case report. J Med Case Rep. 2020;14(1):187.
Tholin, B., Hauge, M. T., Aukrust, P., Fehrle, L., & Tvedt, T. H. (2020). Hemophagocytic lymphohistiocytosis in a patient with COVID-19 treated with tocilizumab: a case report. Journal of Medical Case Reports, 14(1), 187. https://doi.org/10.1186/s13256-020-02503-9
Tholin B, et al. Hemophagocytic Lymphohistiocytosis in a Patient With COVID-19 Treated With Tocilizumab: a Case Report. J Med Case Rep. 2020 Oct 15;14(1):187. PubMed PMID: 33054818.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Hemophagocytic lymphohistiocytosis in a patient with COVID-19 treated with tocilizumab: a case report. AU - Tholin,Birgitte, AU - Hauge,Marit Teigen, AU - Aukrust,Pål, AU - Fehrle,Lutz, AU - Tvedt,Tor Henrik, Y1 - 2020/10/15/ PY - 2020/05/08/received PY - 2020/08/17/accepted PY - 2020/10/15/entrez PY - 2020/10/16/pubmed PY - 2020/11/11/medline KW - COVID-19 KW - Case report KW - HLH KW - Hemophagocytic lymphohistiocytosis KW - IL-6 KW - Tocilizumab SP - 187 EP - 187 JF - Journal of medical case reports JO - J Med Case Rep VL - 14 IS - 1 N2 - BACKGROUND: The understanding of coronavirus disease 2019 (COVID-19) is rapidly evolving. Although it is primarily a respiratory illness, other manifestations, such as Guillain-Barré syndrome, immune thrombocytopenia, and immune-mediated thrombotic thrombocytopenic purpura, have been described. We present a case of a patient with hemophagocytic lymphohistiocytosis secondary to COVID-19 treated with tocilizumab with a marked biochemical improvement. CASE PRESENTATION: In this case report we present a Caucasian patient with COVID-19 who developed a marked elevation of inflammatory parameters with ferritin 36,023 μg/L, but also elevated C-reactive protein 334 mg/L and lactate dehydrogenase 1074 U/L, 1 week after admission to the intensive care unit. He met five of eight criteria for hemophagocytic lymphohistiocytosis, but he lacked the high fever and cytopenia seen in the majority of cases. He was treated with tocilizumab, a monoclonal antibody targeting the interleukin-6 receptor, and over the next days, a rapid decrease in ferritin and C-reactive protein levels was observed. However, his respiratory failure only improved gradually, and he was weaned off the respirator 11 days later. CONCLUSION: COVID-19 may induce a hyperinflammatory clinical picture and in some cases develop into hemophagocytic lymphohistiocytosis. In our patient's case, therapeutic interleukin-6 blockade abrogated signs of hyperinflammation but did not seem to improve pulmonary function. Measurement of ferritin and C-reactive protein, as well as quantification of interleukin-6 on indication, should be performed in patients with severe COVID-19. Specific treatment in such patients must also be contemplated, preferably in randomized controlled trials. SN - 1752-1947 UR - https://www.unboundmedicine.com/medline/citation/33054818/Hemophagocytic_lymphohistiocytosis_in_a_patient_with_COVID_19_treated_with_tocilizumab:_a_case_report_ DB - PRIME DP - Unbound Medicine ER -