J Neurol (2014) 261:432–434 DOI 10.1007/s00415-013-7213-7

LETTER TO THE EDITORS

Toothbrushing-induced seizures at onset of cryptogenic partial epilepsy: a case report Derek V. M. Ott • Andreas Kauert Martin Holtkamp



Received: 30 November 2013 / Revised: 5 December 2013 / Accepted: 7 December 2013 / Published online: 25 December 2013 Ó Springer-Verlag Berlin Heidelberg 2013

Dear sirs, A 47-year-old patient suffered recurring impairment of consciousness, lasting for 20 s to 2 min, every few days when brushing his teeth. Mental relaxation (e.g. on weekends or after sexual intercourse) was observed to favor these events. At first neurological consulting, the series had been going on for 1 year; he reported approximately ten episodes per month, two-thirds whereof on weekends. A home video recorded by his wife shows him standing at the sink, eyes wide open, salivating heavily, smacking, and performing rocking or brief jerky movements. At most, he replies with an inadequate murmur or shrug when spoken to. After approximately 2 min, he gradually regains consciousness and responds coherently, but remains amnesic for the episode itself. Personal medical or family history was inconspicuous; neurological exam, 3 T brain MRI, and cerebrospinal fluid analyses (including anti-neural antibodies) resulted normal. Repeated toothbrushing during 48 h video-EEG-monitoring, however, lead to a total of three complex partial seizures with the above-described semiology that lasted for

Electronic supplementary material The online version of this article (doi:10.1007/s00415-013-7213-7) contains supplementary material, which is available to authorized users. D. V. M. Ott (&)  M. Holtkamp Epilepsy-Center Berlin-Brandenburg, Evangelisches Krankenhaus Ko¨nigin Elisabeth Herzberge, Herzbergstr. 79, 10365 Berlin, Germany e-mail: [email protected] A. Kauert Department of Neurology, Evangelisches Krankenhaus Ko¨nigin Elisabeth Herzberge, Berlin, Germany

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approximately 2 min and were accompanied by a left or non-lateralized EEG seizure pattern (Fig. 1). We reasoned that antiepileptic drugs might be avoided by modification of the oral trigger stimulus and recommended the use of an electric toothbrush, applied with slight pressure only. Indeed, the patient reported at followup that the powered toothbrush did not induce seizures. Unfortunately, however, unprovoked seizures began to occur at arbitrary time points; therefore, he had returned to using his manual toothbrush, hoping to regain control over seizures occurrence. To his disappointment, spontaneous seizures persisted after only three successful attempts of deliberate triggering. We therefore began treatment with levetiracetam, gradually raising the daily dose to 3,000 mg; however, seizure frequency increased to an almost daily basis. We added lacosamide, which at 200 mg per day resulted in freedom from seizures for 3 months at last follow-up. Epileptic seizures induced by toothbrushing are very rare and have so far been described in nine patients only [1–7]. They are regarded as a form of reflex epilepsy, where seizures are precipitated by either simple sensory stimuli (e.g. photosensitivity) or higher order tasks (e.g. primary reading epilepsy) [8]. Since toothbrushing inseparably involves repetitive hand movement, tonic activity of facial muscles, and sensory feedback, the proconvulsive effect normally cannot be assigned to a single modality and may as well be synergistic. An interesting exception is the report of a severely disabled girl who had her teeth brushed [7], suggesting a sensory trigger in this case. Most patients additionally suffered seizures not linked to toothbrushing itself (or thinking of or seeing a toothbrush) [4], but unprovoked [1, 4, 7] or dependent on other oral triggers such as eating [5]. Semiology of seizures, overall neurological status, EEG, and brain MRI findings, and type and

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Fig. 1 Ictal EEG of a seizure provoked by toothbrushing shows a left-lateralized central seizure pattern at onset (circle).

success of therapeutic intervention are highly heterogeneous across reports (see Online Supplementary Material), rendering a common syndrome rather improbable. Of particular interest here, however, is the case of a patient with left temporal lobe epilepsy who had been seizure-free under medication but developed auras during toothbrushing and nocturnal generalized tonic–clonic seizures the moment she started using a powered toothbrush. Seizure activity came to a sudden stop when she switched back to a manual toothbrush [6]. In the present report, the patient’s epilepsy showed a remarkably rapid deterioration from complete health to approximately ten toothbrushing-induced seizures per month, subsequently evolving into cryptogenic partial epilepsy with frequent unprovoked seizures after changing toothbrushing behavior. Antiepileptic polytherapy with levetiracetam and lacosamide effectuated sudden seizure-freedom. The etiology of this reflex epilepsy and its course towards unprovoked and difficult-to-treat seizures remain enigmatic. This case adds to the small and noticeably

heterogeneous series of patients who only share seizure precipitation by toothbrushing. Conflicts of interest D. Ott and A. Kauert report no disclosures. M. Holtkamp has served on scientific advisory boards for UCB, Eisai Inc., and Janssen. He has received reimbursement for travelling expenses and/or speaker honoraria from UCB, Eisai Inc., Desitin, Janssen, GlaxoSmithKline, and Viropharm. Ethical standard We confirm that we have read the journal’s position on issues involved in ethical publication and confirm that this report is consistent with the guidelines.

References 1. Holmes GL, Blair S, Eisenberg E, Scheebaum R, Margraf J, Zimmerman AW (1982) Tooth-brushing-induced epilepsy. Epilepsia 23:657–661 2. Koutroumanidis M, Pearce R, Sadoh DR, Panayiotopoulos CP (2001) Tooth brushing-induced seizures: a case report. Epilepsia 42:686–688

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434 3. Chuang YC, Lin TK, Lui CC, Chen SD, Chang CS (2004) Toothbrushing epilepsy with ictal orgasms. Seizure 13:179–182 4. Navarro V, Adam C, Petitmengin C, Baulac M (2006) Toothbrushthinking seizures. Epilepsia 47:1971–1973 5. D’Souza WJ, O’Brien TJ, Murphy M, Trost NM, Cook MJ (2007) Toothbrushing-induced epilepsy with structural lesions in the primary somatosensory area. Neurology 68:769–771 6. Haytac MC, Aslan K, Ozcelik O, Bozdemir H (2008) Epileptic seizures triggered by the use of a powered toothbrush. Seizure 17:288–291

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J Neurol (2014) 261:432–434 7. Kumada T, Nishii R, Higashi T et al (2013) Passive toothbrushinginduced seizures: report of a severely disabled girl. Brain Dev 35:91–94 8. Wolf P, Koepp M (2012) Reflex epilepsies. Handb Clin Neurol 107:257–276

Toothbrushing-induced seizures at onset of cryptogenic partial epilepsy: a case report.

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