Active neuroborreliosis or inflammation: are the diagnostic guidelines at stake?

  • Harald De CauwerEmail author
  • Katrien Lagrou
  • Evelien Coeckelbergh
  • Ann Bogaerts
  • Jo Leenders
  • Dieter Vanneste
Original article


Neuroborreliosis can induce a variety of neurological syndromes: e.g., cranial neuritis, plexitis, radiculitis, meningitis, cerebellitis, … We report on five cases of patients with a diagnosis of neuroborreliosis based on clinical symptoms, serologic tests and MR imaging results. However, neither of them fulfils the diagnostic criteria for definite neuroborreliosis. Are the diagnostic criteria still valid or is there a need to revise them? Is our diagnosis correct? Are these cases post-Lyme auto-immune neuronal inflammation, and not due to still active spirochetal infection? Do we need to consider immunosuppressive therapy instead of third-generation cephalosporins?


Neuroborreliosis Differential diagnosis Myelitis Diagnostic criteria MR imaging. 


Compliance with ethical standards

Conflict of interest

All the authors report no disclosure or conflict of interest relevant to the manuscript.

Ethical approval

This manuscript does not contain any studies with human participants or animals performed by any of the authors.

Informed consent

Informed consent was obtained from all individual participants included in the study. Additional informed consent was obtained from all individual participants for whom identifying information is included in this article.


  1. 1.
    Belgische commissie voor de coördinatie van het antibioticabeleid (BAPCOC). Aanbevelingen BAPCOC Lyme borreliose 2017. Accessed January 12, 2018
  2. 2.
    Bleyenheuft C, Lernout T, Berger N, Rebolledo J, Leroy M, Robert A, Quoilin S (2015) Epidemiological situation of Lyme borreliosis in Belgium, 2003 to 2012. Arch Public Health 73(1):33CrossRefGoogle Scholar
  3. 3.
    Obsomer V, Wirtgen M, Linden A, Claerebout E, Heyman P, Heylen D, Madder M, Maris J, Lebrun M, Tack W, Lempereur L, Hance T, Van Impe G (2013) Spatial disaggregation of tick occurrence and ecology at a local scale as a preliminary step for spatial surveillance of tick-borne diseases: general framework and health implications in Belgium. Parasit Vectors 22;6:190CrossRefGoogle Scholar
  4. 4.
    Linard C, Tersago K, Vanwambeke S, Lambin E (2007) Determinants of the geographic distribution of Puumala virus and Lyme borreliosis infections in Belgium. International Journal of Health Geographics 6:15CrossRefGoogle Scholar
  5. 5.
    De Keukeleire M, Robert A, Kabamba B, Dion E, Luyasu V, Vanwambeke SO (2016) Individual and environmental factors associated with the seroprevalence of Borrelia burgdorferi in Belgian farmers and veterinarians. Infect Ecol Epidemiol 6:32793CrossRefGoogle Scholar
  6. 6.
    De Keukeleire M, Vanwambeke SO, Somassè E, Kabamba B, Luyasu V, Robert A (2015) Scouts, forests, and ticks: Impact of landscapes on human-tick contacts. Ticks Tick Borne Dis 6(5):636–644CrossRefGoogle Scholar
  7. 7.
    Kesteman T, Rossi C, Bastien P, Brouillard J, Avesani V, Olive N, Martin P, Delmée M (2010) Prevalence and genetic heterogeneity of Borrelia burgdorferi sensu lato in Ixodes ticks in Belgium. Acta Clin Belg 65(5):319–322CrossRefGoogle Scholar
  8. 8.
    De Keukeleire M, Vanwambeke S, Kabamba B, Belkhir L, Pierre P, Luyasu V, Robert A (2017) Time trend of clinical cases of Lyme disease in two hospitals in Belgium, 2000–2013. BMC Infect Dis 17:748CrossRefGoogle Scholar
  9. 9.
    Jungnick S, Margos G, Rieger M, Dzaferovic E, Bent SJ, Overzier E et al (2015) Borrelia burgdorferi sensu stricto and Borrelia afzelii: population structure and differential pathogenicity. Int J Med Microbiol 305:673–681CrossRefGoogle Scholar
  10. 10.
    Cerar T, Strle F, Stupica D, Ruzic-Sabljic E, McHugh G, Steere A, Strle K (2016) Differences in Genotype, Clinical Features, and Inflammatory Potential of Borrelia burgdorferi sensu stricto Strains from Europe and the United States. Emerg Infect Dis 22(5):818–827CrossRefGoogle Scholar
  11. 11.
    Hengge UR, Tannapfel A, Tyring SK, Erbel R, Arendt G, Ruzicka T (2003) Lyme Borreliosis. Lancet Infect Dis 3:489–500CrossRefGoogle Scholar
  12. 12.
    Hildenbrand P, Craven DE, Jones R, Nemeskai P (2009) Lyme Neuroborreliosis: manifestations of a rapidly emerging zoonosis. Am J Neuroradiol 30:1079–1987CrossRefGoogle Scholar
  13. 13.
    Kříž B, Malý M, Daniel M (2017) Neuroborreliosis in patients hospitalised for Lyme borreliosis in the Czech Republic in 2003–2013. Epidemiol Mikrobiol Imunol 66(3):115–123Google Scholar
  14. 14.
    Mygland A, Ljøstad U, Fingerle V, Rupprecht T, Schmutzhard E, Steiner I (2010) European Federation of Neurological Societies. EFNS guidelines on the diagnosis and management of European Lyme neuroborreliosis. Eur J Neurol 17(1):8–16, e1-4CrossRefGoogle Scholar
  15. 15.
    Stanek G, Fingerle V, Hunfeld KP, Jaulhac B, Kaiser R, Krause A, Kristoferitsch W, O’Connell S, Ornstein K, Strle F, Gray J (2011) Lyme borreliosis: clinical case definitions for diagnosis and management in Europe. Clin Microbiol Infect 17(1):69–79CrossRefGoogle Scholar
  16. 16.
    Dessau RB, van Dam AP, Fingerle V, Gray J, Hovius JW, Hunfeld KP, Jaulhac B, Kahl O, Kristoferitsch W, Lindgren PE, Markowicz M, Mavin S, Ornstein K, Rupprecht T, Stanek G, Strle F. To test or not to test? Laboratory support for the diagnosis of Lyme borreliosis: a position paper of ESGBOR, the ESCMID study group for Lyme borreliosis. Clin Microbiol Infect. 2017. pii: S1198-743X(17)30488-3Google Scholar
  17. 17.
    Belman AL, Reynolds L, Preston T, Postels D, Grimson R, Coyle PK (1997) Cerebrospinal fluid findings in children with Lyme disease-associated facial nerve palsy. Arch Pediatr Adolesc Med 151(12):1224–1228CrossRefGoogle Scholar
  18. 18.
    Tumani H, Nölker G, Reiber H (1995) Relevance of cerebrospinal fluid variables for early diagnosis of neuroborreliosis. Neurology 45(9):1663–1670CrossRefGoogle Scholar
  19. 19.
    Djukic M, Schmidt-Samoa C, Lange P, Spreer A, Neubieser K, Eiffert H, Nau R, Schmidt H (2012) Cerebrospinal fluid findings in adults with acute Lyme neuroborreliosis. J Neurol 259(4):630–636CrossRefGoogle Scholar
  20. 20.
    Ljøstad U, Skarpaas T, Mygland A (2007) Clinical usefulness of intrathecal antibody testing in acute Lyme neuroborreliosis. Eur J Neurol 14(8):873–876CrossRefGoogle Scholar
  21. 21.
    Eckman EA, Pacheco-Quinto J, Herdt AR, Halperin JJ (2018) Neuroimmunomodulators in neuroborreliosis and Lyme encephalopathy. Clin Infect Dis. Google Scholar
  22. 22.
    De Cauwer H, Declerck S, De Smet J, Matthyssen Ph, Pelzers E, Eykens L (2009) K. Lagrou. Motor neuron disease features in a patient with neuroborreliosis. Acta clinica belgica 64:225–227CrossRefGoogle Scholar
  23. 23.
    Charles V, Duprez TP, Kabamba B, Ivanoiu A, Sindic CJM (2007) Poliomyelitis-like syndrome with matching magnetic resonance features in a case of Lyme neuroborreliosis. J Neurol Neurosurg Psychiatry 78:1160–1161CrossRefGoogle Scholar
  24. 24.
    Siao Tick Chong P, Vucic S, Cros DP (2003) Multiple sclerosis presenting as lower motor neuron wasting and weakness of the distal upper extremity. Neurology 61(9):1303–1304CrossRefGoogle Scholar
  25. 25.
    Lodewyckx AA, De Cauwer H (2015) Intramedullaire neurosarcoïdose: casusbespreking en overzicht van de literatuur. Tijdschrift voor geneeskunde 71(8):527–532Google Scholar
  26. 26.
    Nardone R, Versace V, Brigo F, Tezzon F, Zuccoli G, Pikija S, Hauer L, Sellner J (2017) Herpes Simplex Virus Type 2 Myelitis: Case Report and Review of the Literature. Front Neurol 8:199CrossRefGoogle Scholar
  27. 27.
    Ramesh G, Didier PJ, England JD, Santana-Gould L, Doyle-Meyers LA, Martin DS, Jacobs MB, Philipp MT (2015) Inflammation in the pathogenesis of lyme neuroborreliosis. Am J Pathol 185(5):1344–1360CrossRefGoogle Scholar
  28. 28.
    Embers ME, Hasenkampf NR et al (2017) Variable manifestations, diverse seroreactivity and post-treatment persistence in non-human primates exposed to Borrelia burgdorferi by tick feeding. PLoS One 12(12):e0189071CrossRefGoogle Scholar
  29. 29.
    Crossland NA, Alvarez X, Embers ME. Late Disseminated Lyme Disease: Associated Pathology and Spirochete Persistence Post-Treatment in Rhesus Macaques. Am J Pathol. 2017Google Scholar
  30. 30.
    Rothstein JD, Patel S, Regan MR, Haenggeli C, Huang YH, Bergles DE, Jin L, Dykes Hoberg M, Vidensky S, Chung DS, Toan SV, Bruijn LI, Su ZZ, Gupta P, Fisher PB (2005) Beta-lactam antibiotics offer neuroprotection by increasing glutamate transporter expression. Nature 433(7021):73–77CrossRefGoogle Scholar
  31. 31.
    Hsieh MH, Meng WY, Liao WC, Weng JC, Li HH, Su HL, Lin CL, Hung CS, Ho YJ (2017) Ceftriaxone reverses deficits of behavior and neurogenesis in an MPTP-induced rat model of Parkinson’s disease dementia. Brain Res Bull 132:129–138CrossRefGoogle Scholar
  32. 32.
    Krzyzanowska W, Pomierny B, Budziszewska B, Filip M, Pera J (2016) N-Acetylcysteine and Ceftriaxone as Preconditioning Strategies in Focal Brain Ischemia: Influence on Glutamate Transporters Expression. Neurotox Res 29(4):539–550CrossRefGoogle Scholar
  33. 33.
    Cui C, Cui Y, Gao J, Sun L, Wang Y, Wang K, Li R, Tian Y, Song S, Cui J (2014) Neuroprotective effect of ceftriaxone in a rat model of traumatic brain injury. Neurol Sci 35(5):695–700CrossRefGoogle Scholar
  34. 34.
    Jeannet N, van den Hoogen BG, Schefold JC, Suter-Riniker F, Sommerstein R (2017) Cerebrospinal Fluid Findings in an Adult with Human Metapneumovirus-Associated Encephalitis. Emerg Infect Dis 23(2):370CrossRefGoogle Scholar
  35. 35.
    Mohammad SS, Dale RC (2018) Principles and approaches to the treatment of immune-mediated movement disorders. Eur J Paediatr Neurol 22(2):292–300CrossRefGoogle Scholar
  36. 36.
    Akins PT, Belko J, Uyeki TM, Axelrod Y, Lee KK, Silverthorn J (2010) H1N1 encephalitis with malignant edema and review of neurologic complications from influenza. Neurocrit Care 13(3):396–406CrossRefGoogle Scholar

Copyright information

© Belgian Neurological Society 2018

Authors and Affiliations

  1. 1.Department of NeurologyAZ St DimpnaGeelBelgium
  2. 2.Faculty of Medicine and Health SciencesUniversity of AntwerpWilrijkBelgium
  3. 3.Department of Microbiology and Immunology, Laboratory of Clinical Bacteriology and MycologyKU Leuven-University of LeuvenLeuvenBelgium
  4. 4.Clinical Laboratory, Laboratory of MicrobiologyAZ St DimpnaGeelBelgium
  5. 5.Department of NeuroradiologyAZ St DimpnaGeelBelgium
  6. 6.Department of NeuroradiologyUniversity Hospital GasthuisbergLeuvenBelgium

Personalised recommendations