Highligts
- •First description of Cerebrospinal fluid changes in BoDV-1 encephalitis.
- •CSF white blood cell count was increased in first examination in 15 of 18 cases and completely normal CSF is uncommon.
- •CSF changes seem similar to encephalitis due to other viruses.
- •BoDV-1 PCR may remain negative in the CSF.
- •Four of five patients tested showed a polyvalent reaction against multiple viruses in the CSF suggesting that BoDV1 may trigger autoimmune mechanisms.
Abstract
Borna disease virus 1 (BoDV-1) has been recognized as a rare cause of very severe
encephalitis with rapid onset in central Europe. Data on cerebrospinal fluid (CSF)
analysis have not yet been analyzed in detail. Here, we present the first study on
CSF changes in BoDV-1 encephalitis.
We retrospectively analyzed CSFs from 18 BoDV-1 encephalitis cases from Bavaria, Germany,
an endemic region, from 1996 to 2021. Data were obtained through review of medical
records and institutional databases.
We found that white blood cell count (WBC) in CSF is elevated in 13 of our 18 patients
at first examination (average 83.2 ± 142.3 leukocytes/μl) and cytology showed predominance
of lymphocytes. Patients with typical symptoms of meningoencephalitis had higher WBC
in first CSF analyzation (133.5 ± 163.1 vs 4.0 ± 3.2/μl; p = 0.065). BoDV-1 PCR of CSF is not always positive when tested (7 of 9 cases). Four
of five patients tested showed a polyvalent reaction against multiple viruses in the
CSF suggesting that BoDV1 may trigger autoimmune mechanisms.
CSF changes in BoDV-1 encephalitis seem similar to those of other viral encephalitis
and at the beginning WBC can be normal in up to 28%, making the diagnosis even more
challenging. All in all, BoDV-1 should be included in the diagnostic workup of patients
with rapidly evolving and/or severe encephalitis and patients with severe neuropathy
and secondary encephalopathy with and without CSF changes. Repeated CSF examinations
as well as BoDV-1 serology and CSF PCR have to be considered in endemic areas.
Keywords
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References
- Fatal encephalitic Borna disease virus 1 in solid-organ transplant recipients.N. Engl. J. Med. 2018; 379 (PMID: 30281984): 1377-1379https://doi.org/10.1056/NEJMc1803115
- Zoonotic spillover infections with Borna disease virus 1 leading to fatal human encephalitis, 1999-2019: an epidemiological investigation.Lancet Infect. Dis. 2020; 20 (Epub 2020 Jan 7. PMID: 31924550): 467-477https://doi.org/10.1016/S1473-3099(19)30546-8
Liesche F, Ruf V, Zoubaa S, Kaletka G, Rosati M, Rubbenstroth D, Herden C, Goehring L, Wunderlich S, Wachter MF, Rieder G, Lichtmannegger I, Permanetter W, Heckmann JG, Angstwurm K, Neumann B, Märkl B, Haschka S, Niller HH, Schmidt B, Jantsch J, Brochhausen C, Schlottau K, Ebinger A, Hemmer B, Riemenschneider MJ, Herms J, Beer M, Matiasek K, Schlegel J. The neuropathology of fatal encephalomyelitis in human Borna virus infection. Acta Neuropathol. 2019;138(4):653–665. doi: https://doi.org/10.1007/s00401-019-02047-3. (Epub 2019 Jul 26. PMID: 31346692; PMCID: PMC6778062)
- Bornavirus encephalitis shows a characteristic magnetic resonance phenotype in humans.Ann. Neurol. 2020; 88 (Epub 2020 Aug 29. PMID: 32794235): 723-735https://doi.org/10.1002/ana.25873
- Antibodies against viral nucleo-, phospho-, and X protein contribute to serological diagnosis of fatal Borna disease virus 1 infections.Cell Rep Med. 2022; 3 (PMID: 35106511; PMCID: PMC8784767)100499https://doi.org/10.1016/j.xcrm.2021.100499
- Fatal encephalitis associated with Borna disease virus 1.N. Engl. J. Med. 2018; 379 (PMID: 30281979): 1375-1377https://doi.org/10.1056/NEJMc1800724
- Die Borna-Virus-Enzephalitis als Differenzialdiagnose zur seronegativen Autoimmunenzephalitis [Bornavirus encephalitis as a differential diagnosis to seronegative autoimmune encephalitis].Nervenarzt. 2022; (German) (Epub ahead of print. Erratum in: Nervenarzt. 2022 Apr 22;: PMID: 35024881; PMCID: PMC8756745): 1-3
- Comparison of laboratory and immune characteristics of the initial and second phase of tick-borne encephalitis.Emerg Microbes Infect. 2022; 11 (PMID: 35657098; PMCID: PMC9225760): 1647-1656https://doi.org/10.1080/22221751.2022.2086070
- Central nervous system infections in the absence of cerebrospinal fluid pleocytosis.Int. J. Infect. Dis. 2017; 65 (Epub 2017 Nov 10. PMID: 29081366): 107-109https://doi.org/10.1016/j.ijid.2017.10.011
- Results of a multinational study suggest the need for rapid diagnosis and early antiviral treatment at the onset of herpetic meningoencephalitis.Antimicrob. Agents Chemother. 2015; 59 (Epub 2015 Mar 16. PMID: 25779579; PMCID: PMC4432209): 3084-3089https://doi.org/10.1128/AAC.05016-14
- Tick-borne encephalitis: a differential pattern of intrathecal humoral immune response and inflammatory cell composition compared with other viral CNS infections.Cells. 2020; 9 (PMID: 32992967; PMCID: PMC7599799): 2169https://doi.org/10.3390/cells9102169
- Antiviral activity of favipiravir (T-705) against mammalian and avian bornaviruses.Antivir. Res. 2017; 143 (Epub 2017 Apr 29. PMID: 28465146): 237-245https://doi.org/10.1016/j.antiviral.2017.04.018
Article info
Publication history
Accepted:
January 22,
2023
Received:
November 9,
2022
Publication stage
In Press Journal Pre-ProofIdentification
Copyright
© 2023 Elsevier B.V. All rights reserved.
