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BMC Neurology
Published in: BMC Neurology 1/2024

Open Access 01-12-2024 | Thrombocytopenia | Case Report

Severe fever with thrombocytopenia syndrome with central nervous system symptom onset: a case report and literature review

Authors: Dawei Shan, Weibi Chen, Gang Liu, Huimin Zhang, Shuting Chai, Yan Zhang

Published in: BMC Neurology | Issue 1/2024

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Abstract

Background

Severe fever with thrombocytopenia syndrome (SFTS) is a natural focal disease transmitted mainly by tick bites, and the causative agent is SFTS virus (SFTSV). SFTS can rapidly progress to severe disease, with multiple-organ failure (MOF) manifestations such as shock, respiratory failure, disseminated intravascular coagulation (DIC) and death, but cases of SFTS patients with central nervous system (CNS) symptoms onset and marked persistent involuntary shaking of the perioral area and limbs have rarely been reported.

Case presentation

A 69-year-old woman with fever and persistent involuntary shaking of the perioral area and limbs was diagnosed with SFTS with CNS symptom onset after metagenomic next-generation sequencing (mNGS) of cerebrospinal fluid (CSF) and peripheral blood identified SFTSV. The patient developed a cytokine storm and MOF during the course of the disease, and after aggressive antiviral, glucocorticoid, and gamma globulin treatments, her clinical symptoms improved, her laboratory indices returned to normal, and she had a good prognosis.

Conclusion

This case gives us great insight that when patients with CNS symptoms similar to those of viral encephalitis combined with thrombocytopenia and leukopenia are encountered in the clinic, it is necessary to consider the possibility of SFTS involving the CNS. Testing for SFTSV nucleic acid in CSF and blood (mNGS or polymerase chain reaction (PCR)) should be carried out, especially in critically ill patients, and treatment should be given accordingly.
Appendix
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Literature
1.
Yu XJ, Liang MF, Zhang SY, Liu Y, Li JD, Sun YL, et al. Fever with thrombocytopenia associated with a novel bunyavirus in China. N Engl J Med. 2011;364(16):1523–32.CrossRefPubMedPubMedCentral
2.
Yang T, Huang H, Jiang L, Li J. Overview of the immunological mechanism underlying severe fever with thrombocytopenia syndrome (review). Int J Mol Med. 2022;50(3).
3.
Li H, Lu QB, Xing B, Zhang SF, Liu K, Du J, et al. Epidemiological and clinical features of laboratory-diagnosed severe fever with thrombocytopenia syndrome in China, 2011-17: a prospective observational study. Lancet Infect Dis. 2018;18(10):1127–37.CrossRefPubMed
4.
Casel MA, Park SJ, Choi YK. Severe fever with thrombocytopenia syndrome virus: emerging novel phlebovirus and their control strategy. Exp Mol Med. 2021;53(5):713–22.CrossRefPubMedPubMedCentral
5.
Fei X, Feng B, Fang K, Ren W. Risk factors for mortality in severe fever with Thrombocytopenia Syndrome patients with Central Nervous System complications. Med Sci Monit. 2023;29:e938427.CrossRefPubMedPubMedCentral
6.
Kobayashi Y, Kato H, Yamagishi T, Shimada T, Matsui T, Yoshikawa T, et al. Severe fever with Thrombocytopenia Syndrome, Japan, 2013–2017. Emerg Infect Dis. 2020;26(4):692–9.CrossRefPubMedPubMedCentral
7.
Park SJ, Kim YI, Park A, Kwon HI, Kim EH, Si YJ, et al. Ferret animal model of severe fever with thrombocytopenia syndrome phlebovirus for human lethal infection and pathogenesis. Nat Microbiol. 2019;4(3):438–46.CrossRefPubMed
8.
Kaneko M, Shikata H, Matsukage S, Maruta M, Shinomiya H, Suzuki T, et al. A patient with severe fever with thrombocytopenia syndrome and hemophagocytic lymphohistiocytosis-associated involvement of the central nervous system. J Infect Chemother. 2018;24(4):292–7.CrossRefPubMed
9.
Guang C, Tao C, Sainan S, Ke M, Xiaojing W, Di W et al. Expert consensus on diagnosis and treatment of severe fever with thrombocytopenia syndrome. Chin J Clin Infect Dis. 2022:253–63.
10.
Park SY, Kwon JS, Kim JY, Kim SM, Jang YR, Kim MC, et al. Severe fever with thrombocytopenia syndrome-associated encephalopathy/encephalitis. Clin Microbiol Infect. 2018;24(4):432.e1-.e4.CrossRef
11.
Cui N, Liu R, Lu QB, Wang LY, Qin SL, Yang ZD, et al. Severe fever with thrombocytopenia syndrome bunyavirus-related human encephalitis. J Infect. 2015;70(1):52–9.CrossRefPubMed
12.
Sun Y, Jin C, Zhan F, Wang X, Liang M, Zhang Q, et al. Host cytokine storm is associated with disease severity of severe fever with thrombocytopenia syndrome. J Infect Dis. 2012;206(7):1085–94.CrossRefPubMed
13.
Deng B, Zhang S, Geng Y, Zhang Y, Wang Y, Yao W, et al. Cytokine and chemokine levels in patients with severe fever with thrombocytopenia syndrome virus. PLoS ONE. 2012;7(7):e41365.CrossRefPubMedPubMedCentral
14.
Kwon JS, Kim MC, Kim JY, Jeon NY, Ryu BH, Hong J, et al. Kinetics of viral load and cytokines in severe fever with thrombocytopenia syndrome. J Clin Virol. 2018;101:57–62.CrossRefPubMedPubMedCentral
15.
Liu MM, Lei XY, Yu H, Zhang JZ, Yu XJ. Correlation of cytokine level with the severity of severe fever with thrombocytopenia syndrome. Virol J. 2017;14(1):6.CrossRefPubMedPubMedCentral
16.
Li J, Li S, Yang L, Cao P, Lu J. Severe fever with thrombocytopenia syndrome virus: a highly lethal bunyavirus. Crit Rev Microbiol. 2021;47(1):112–25.CrossRefPubMed
17.
Fei X, Fang K, Ni X, Ren WH. Risk factors of neurological complications in severe fever patients with thrombolytic syndrome: a single-Center Retrospective Study in China. Med Sci Monit. 2021;27:e932836.CrossRefPubMedPubMedCentral
18.
19.
Li DX. Severe fever with thrombocytopenia syndrome: a newly discovered emerging infectious disease. Clin Microbiol Infect. 2015;21(7):614–20.CrossRefPubMed
20.
Deng B, Zhou B, Zhang S, Zhu Y, Han L, Geng Y, et al. Clinical features and factors associated with severity and fatality among patients with severe fever with thrombocytopenia syndrome Bunyavirus infection in Northeast China. PLoS ONE. 2013;8(11):e80802.CrossRefPubMedPubMedCentral
21.
22.
Li H, Zhang LK, Li SF, Zhang SF, Wan WW, Zhang YL, et al. Calcium channel blockers reduce severe fever with thrombocytopenia syndrome virus (SFTSV) related fatality. Cell Res. 2019;29(9):739–53.CrossRefPubMedPubMedCentral
23.
Hayden A, Park S, Giustini D, Lee AY, Chen LY. Hemophagocytic syndromes (HPSs) including hemophagocytic lymphohistiocytosis (HLH) in adults: a systematic scoping review. Blood Rev. 2016;30(6):411–20.CrossRefPubMed
24.
Nakamura S, Azuma M, Maruhashi T, Sogabe K, Sumitani R, Uemura M, et al. Steroid pulse therapy in patients with encephalopathy associated with severe fever with thrombocytopenia syndrome. J Infect Chemother. 2018;24(5):389–92.CrossRefPubMed
25.
Tzekova N, Heinen A, Bunk S, Hermann C, Hartung HP, Reipert B, et al. Immunoglobulins stimulate cultured Schwann cell maturation and promote their potential to induce axonal outgrowth. J Neuroinflammation. 2015;12:107.CrossRefPubMedPubMedCentral
26.
Kim UJ, Kim DM, Ahn JH, Kang SJ, Jang HC, Park KH, et al. Successful treatment of rapidly progressing severe fever with thrombocytopenia syndrome with neurological complications using intravenous immunoglobulin and corticosteroid. Antivir Ther. 2016;21(7):637–40.CrossRefPubMed
27.
Choi S, Kim MC, Kwon JS, Kim JY, Lee KH, Kim SH. Case Report: use of plasma Exchange followed by Convalescent Plasma Therapy in a critically ill patient with severe fever and Thrombocytopenia Syndrome-Associated Encephalopathy: Cytokine/Chemokine concentrations, viral loads, and antibody responses. Am J Trop Med Hyg. 2018;99(6):1466–8.CrossRefPubMedPubMedCentral
28.
Park SY, Choi W, Chong YP, Park SW, Wang EB, Lee WJ, et al. Use of plasma therapy for severe fever with Thrombocytopenia Syndrome Encephalopathy. Emerg Infect Dis. 2016;22(7):1306–8.CrossRefPubMedPubMedCentral
29.
Jia B, Yan X, Chen Y, Wang G, Liu Y, Xu B, et al. A scoring model for predicting prognosis of patients with severe fever with thrombocytopenia syndrome. PLoS Negl Trop Dis. 2017;11(9):e0005909.CrossRefPubMedPubMedCentral
30.
Seo JW, Kim D, Yun N, Kim DM. Clinical update of severe fever with Thrombocytopenia Syndrome. Viruses. 2021;13(7).
31.
Choi SJ, Park SW, Bae IG, Kim SH, Ryu SY, Kim HA, et al. Severe fever with Thrombocytopenia Syndrome in South Korea, 2013–2015. PLoS Negl Trop Dis. 2016;10(12):e0005264.CrossRefPubMedPubMedCentral
32.
Wang L, Wan G, Shen Y, Zhao Z, Lin L, Zhang W, et al. A nomogram to predict mortality in patients with severe fever with thrombocytopenia syndrome at the early stage-A multicenter study in China. PLoS Negl Trop Dis. 2019;13(11):e0007829.CrossRefPubMedPubMedCentral
33.
Gai ZT, Zhang Y, Liang MF, Jin C, Zhang S, Zhu CB, et al. Clinical progress and risk factors for death in severe fever with thrombocytopenia syndrome patients. J Infect Dis. 2012;206(7):1095–102.CrossRefPubMed
34.
Chen Y, Jia B, Liu Y, Huang R, Chen J, Wu C. Risk factors associated with fatality of severe fever with thrombocytopenia syndrome: a meta-analysis. Oncotarget. 2017;8(51):89119–29.CrossRefPubMedPubMedCentral
35.
Yoshikawa T, Fukushi S, Tani H, Fukuma A, Taniguchi S, Toda S, et al. Sensitive and specific PCR systems for detection of both Chinese and Japanese severe fever with thrombocytopenia syndrome virus strains and prediction of patient survival based on viral load. J Clin Microbiol. 2014;52(9):3325–33.CrossRefPubMedPubMedCentral
36.
Saijo M. Pathophysiology of severe fever with thrombocytopenia syndrome and development of specific antiviral therapy. J Infect Chemother. 2018;24(10):773–81.CrossRefPubMed
37.
Youdong X, Xiaofeng D, Xiyuan N, Zhengdong L. Analysis of the risk factors and prognosis for severe fever with thrombocytopenia syndrome associated encephalopathy. J Infect Chemother. 2023;29(5):464–8.CrossRefPubMed
38.
Kawaguchi T, Matsuda M, Takajo I, Kawano A, Kariya Y, Kubo K, et al. Severe fever with thrombocytopenia syndrome with myocardial dysfunction and encephalopathy: a case report. J Infect Chemother. 2016;22(9):633–7.CrossRefPubMed
39.
Kim UJ, Kim DM, Kim SE, Kang SJ, Jang HC, Park KH, et al. Case report: detection of the identical virus in a patient presenting with severe fever with thrombocytopenia syndrome encephalopathy and the tick that bit her. BMC Infect Dis. 2018;18(1):181.CrossRefPubMedPubMedCentral
40.
Sun Y, Guo B, Yan H, Wu AL, Yao WW, Chen K, et al. Patient with severe fever with thrombocytopenia syndrome virus infection and central nervous system disturbance in Dongyang, Zhejiang Province, China, 2017. Virol J. 2019;16(1):129.CrossRefPubMedPubMedCentral
41.
Wang C, Gong L, Zeng Z, Zhang J, Guan H, Chen L, et al. Genome-based analysis of SFTSV causing severe encephalitis with brain lesions. J Neurovirol. 2020;26(2):181–7.CrossRefPubMed
Metadata
Title
Severe fever with thrombocytopenia syndrome with central nervous system symptom onset: a case report and literature review
Authors
Dawei Shan
Weibi Chen
Gang Liu
Huimin Zhang
Shuting Chai
Yan Zhang
Publication date
01-12-2024
Publisher
BioMed Central
Published in
BMC Neurology / Issue 1/2024
Electronic ISSN: 1471-2377
DOI
https://doi.org/10.1186/s12883-024-03664-6

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