epileptic seizure as initial and only manifestation of neuroborreliosis: case report

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LETTER TO THE EDITOR Epileptic seizure as initial and only manifestation of neuroborreliosis: case report Stjepan Juric Davor Janculjak Svetlana Tomic Silva Butkovic Soldo Ervina Bilic Received: 10 December 2013 / Accepted: 17 January 2014 Ó Springer-Verlag Italia 2014 Introduction Lyme borreliosis is a multisystem disease caused by the spirochete Borrelia burgdorferi [1]. Neuroborreliosis may be manifested as Bannwarth’s syndrome (symptom triad: facial palsy, lymphocytic meningitis, and radicular pain), progressive encephalomyelitis, encephalopathy, cerebral vasculitis, transverse myelitis, amyotrophic lateral sclerosis syndrome, internal hydrocephalus, dementia, depression, axonal motor polyneuropathy, acrodermatitis chronica atrophicans, radicular lesions, and sensory neuropathies [1]. We present a patient who developed epileptic seizure as initial and only manifestation of neuroborreliosis. Case report A 46-year-old man was admitted after partial motor epi- leptic seizure with secondary generalization, preceded by general weakness for several days before. The patient had no serious disease, epilepsy, perinatal trauma, head injury, previous CNS inflammatory diseases, or febrile convul- sions in his past medical history. He reported tick bites on several occasions. One year before, he had rash on the abdomen and chest skin, which has cleared spontaneously. When admitted, the patient showed mild right-sided hem- iparesis with motor dysphasia, which was resolved on the same day. He also had reddish-purple rash on the trunk and the abdomen skin that has cleared in several days. Upon admission, he underwent computed tomography (CT) of the brain and fundoscopy. The results were normal. Brain MRI performed on day 2 revealed a demyelination zone of open etiology temporally on the left. Serologic testing for B. burgdorferi was positive (enzyme-linked immunosorbent assay, ELISA: IgM positive and IgG positive). Repeated serology (ELISA and Western blot) was positive at a titer of 1:512. Electroencephalogram (EEG) acquired on the day 2 of the disease showed dys- rhythmic changes, with a focus temporally on the left. CSF testing showed slightly elevated protein concen- tration, with no other pathology. Electrocardiography and cardiologic examination revealed no signs of cardiac impairment. An extensive workup for vasculitides, coagu- lation disorders, hematologic diseases and other infectious diseases produced normal findings. Antiepileptic therapy was introduced (carbamazepine, 600 mg/day) and the patient was transferred to the Department of Infectious Diseases, where he has received antibiotic therapy (ceftriaxone, 2 g intravenous/17 days). The patient did not experience new seizures throughout the 4-year follow-up monitoring by an epileptologist. Two years after the symptomatic epileptic seizure, EEG finding revealed only minor changes. The follow-up MRI of the brain showed normal finding. Therefore, antiepileptic therapy was discontinued. Discussion The clinical presentation and course of the disease in our patient pointed to a symptomatic partial motor seizure with secondary generalization. Brain MRI revealed an area of S. Juric (&) Á D. Janculjak Á S. Tomic Á S. Butkovic Soldo Department of Neurology, University Hospital Center Osijek, J. Huttlera 4, 31000 Osijek, Croatia e-mail: [email protected] E. Bilic Department of Neurology, University Hospital Center Zagreb, Zagreb, Croatia 123 Neurol Sci DOI 10.1007/s10072-014-1648-1

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Page 1: Epileptic seizure as initial and only manifestation of neuroborreliosis: case report

LETTER TO THE EDITOR

Epileptic seizure as initial and only manifestationof neuroborreliosis: case report

Stjepan Juric • Davor Janculjak • Svetlana Tomic •

Silva Butkovic Soldo • Ervina Bilic

Received: 10 December 2013 / Accepted: 17 January 2014

� Springer-Verlag Italia 2014

Introduction

Lyme borreliosis is a multisystem disease caused by the

spirochete Borrelia burgdorferi [1]. Neuroborreliosis may

be manifested as Bannwarth’s syndrome (symptom triad:

facial palsy, lymphocytic meningitis, and radicular pain),

progressive encephalomyelitis, encephalopathy, cerebral

vasculitis, transverse myelitis, amyotrophic lateral sclerosis

syndrome, internal hydrocephalus, dementia, depression,

axonal motor polyneuropathy, acrodermatitis chronica

atrophicans, radicular lesions, and sensory neuropathies [1].

We present a patient who developed epileptic seizure as

initial and only manifestation of neuroborreliosis.

Case report

A 46-year-old man was admitted after partial motor epi-

leptic seizure with secondary generalization, preceded by

general weakness for several days before. The patient had

no serious disease, epilepsy, perinatal trauma, head injury,

previous CNS inflammatory diseases, or febrile convul-

sions in his past medical history. He reported tick bites on

several occasions. One year before, he had rash on the

abdomen and chest skin, which has cleared spontaneously.

When admitted, the patient showed mild right-sided hem-

iparesis with motor dysphasia, which was resolved on the

same day. He also had reddish-purple rash on the trunk and

the abdomen skin that has cleared in several days. Upon

admission, he underwent computed tomography (CT) of

the brain and fundoscopy. The results were normal. Brain

MRI performed on day 2 revealed a demyelination zone of

open etiology temporally on the left.

Serologic testing for B. burgdorferi was positive

(enzyme-linked immunosorbent assay, ELISA: IgM positive

and IgG positive). Repeated serology (ELISA and Western

blot) was positive at a titer of 1:512. Electroencephalogram

(EEG) acquired on the day 2 of the disease showed dys-

rhythmic changes, with a focus temporally on the left.

CSF testing showed slightly elevated protein concen-

tration, with no other pathology. Electrocardiography and

cardiologic examination revealed no signs of cardiac

impairment. An extensive workup for vasculitides, coagu-

lation disorders, hematologic diseases and other infectious

diseases produced normal findings.

Antiepileptic therapy was introduced (carbamazepine,

600 mg/day) and the patient was transferred to the

Department of Infectious Diseases, where he has received

antibiotic therapy (ceftriaxone, 2 g intravenous/17 days).

The patient did not experience new seizures throughout the

4-year follow-up monitoring by an epileptologist. Two

years after the symptomatic epileptic seizure, EEG finding

revealed only minor changes. The follow-up MRI of the

brain showed normal finding. Therefore, antiepileptic

therapy was discontinued.

Discussion

The clinical presentation and course of the disease in our

patient pointed to a symptomatic partial motor seizure with

secondary generalization. Brain MRI revealed an area of

S. Juric (&) � D. Janculjak � S. Tomic � S. Butkovic Soldo

Department of Neurology, University Hospital Center Osijek,

J. Huttlera 4, 31000 Osijek, Croatia

e-mail: [email protected]

E. Bilic

Department of Neurology, University Hospital Center Zagreb,

Zagreb, Croatia

123

Neurol Sci

DOI 10.1007/s10072-014-1648-1

Page 2: Epileptic seizure as initial and only manifestation of neuroborreliosis: case report

nonspecific demyelination, which was responsible for the

epileptiform activity recorded on EEG. Initial and repeated

serologic testing for B. burgdorferi yielded positive results

and we decided to introduce antibiotic therapy.

Bannwarth’s syndrome and disorders associated with

meningoencephalitis, peripheral nervous system lesions,

cranial nerve lesions, and painful syndromes are described

in the literature as the most frequent manifestations of

neuroborreliosis [1]. Epileptic seizure is reported rarely as

neuroborreliosis manifestation, while epileptic seizure as

the only manifestation of neuroborreliosis is reported

exceptionally rarely [2, 3].

The change observed on the brain MRI was probably an

atypical vascular ischemic lesion consequential to the

development of vasculitis [4]. Vasculitis is the primary

pathophysiological mechanism of ischemic lesions in

neuroborreliosis [4] and, therefore, of the consequential

epileptogenic focus and the epileptic seizures. Some liter-

ature reports suggest that neuroborreliosis itself acts as a

trigger of the epileptic seizure [5]. Vasculitis, as a major

cause of CNS symptomatology and epileptic seizures, is

also supported by Mourin et al. They reported a case of a

13-year-old boy with two epileptic seizures and MRI signs

of two brain lesions corresponding to vasculitis. The

authors believe that cerebral vasculitis was the culprit for

these brain lesions and epileptic seizures [2].

A good recovery following early introduction of anti-

biotic therapy has also been described by Staub et al. [6].

This group reported on a 6-year-old boy with the disease

onset manifested by absences and deterioration of the

general somatic condition, headaches and lymphadenopa-

thy, which subsided after 2-week intravenous ceftriaxone

therapy [6].

Conclusion

Epileptic seizure as symptom of neuroborreliosis is less

common, while epileptic seizure as the only manifestation

was rarely reported so far.

References

1. Hansen K, Crone C, Kristoferitsch W (2013) Lyme neuroborre-

liosis. Handb Clin Neurol 115:559–579

2. Mourin S, Bonnier C, Bigaignon G, Lyon G (1993) Epilepsy

disclosing neuroborreliosis. Rev Neurol 149:489–491

3. van Breemen MS, van der Kuip M, Ang CW, van Furth AM, Wolf

NI (2012) Torticollis and seizures due to neuroborreliosis in a

child. Ned Tijdschr Geneeskd 156:A5157

4. Topakian R, Stieglbauer K, Nussbaumer K, Aichner FT (2008)

Cerebral vasculitis and stroke in Lyme neuroborreliosis. Two case

reports and review of current knowledge. Cerebrovasc Dis

26:455–461

5. Baumann M, Birnbacher R, Koch J, Strobl R, Rostasy K (2010)

Uncommon manifestations of neuroborreliosis in children. Eur J

Paediatr Neurol 14:274–277

6. Staub E, Strozzi S, Aebi C (2007) Lymphadenopathy and

absences. Praxis 96:815–817

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