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Reflex Seizures Induced by Micturition and Defecation, Successfully Treated

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Reflex Seizures Induced by Micturition and Defecation, Successfully Treated Original article Epileptic Disord 2011; 13 (2): 166-71 Reflex seizures induced by micturition and defecation, successfully treated with clobazam and phenytoin Tsukasa Higuchi 1,2, Tetsuhiro Fukuyama 1, Yuka Misawa 1, Yuji Inaba 1, Motoki Ichikawa 1, Kenichi Koike 1 1 Department of Pediatrics, Shinshu University School of Medicine, Matsumoto 2 Department of Pediatrics, Nagano Prefectural Suzaka Hospital, Suzaka City, Japan Received February 23, 2010; Accepted February 28, 2011 ABSTRACT – We report a six-year-old girl with seizures induced by both micturition and defecation. Several days after unprovoked generalised tonic-clonic seizures, she developed reflex seizures characterised by the extension of both arms and rhythmic jerking of her upper body. No abnor- mal findings were noted on brain magnetic resonance imaging. Interictal electroencephalography (EEG) showed spike-and-wave activity on central electrode recording, and rhythmic fast activity was recorded by central elec- trodes during the ictal EEG upon micturition. The combination of clobazam and phenytoin was effective for both unprovoked and reflex seizures. Although some previous reports have described reflex seizures triggered by either micturition or defecation, this is the first case report of reflex seizures induced by both micturition and defecation in the same patient. Based on a comparison with previous cases of reflex seizures induced either by micturition or defecation, the neuronal pathway from the pelvic base musculature to the supplementary motor area may be responsible for the condition in our patient. Key words: reflex seizure, micturition, defecation, supplementary motor area, pelvic base musculature, excretion-induced seizures Reflex seizures are evoked by a and defecation are recognized as specific stimulus which is often very rare triggers of reflex seizures. individual to each case (Ritaccio, We describe a case of an epilepsy 1994). Although visual stimuli are patient with reflex seizures caused doi:10.1684/epd.2011.0423 the most frequent triggers of reflex by micturition and defecation, Correspondence: seizures, reading, writing, being based on ictal electroencephalo- T. Higuchi startled, auditory stimuli, immersion graphic findings. The literature on Department of Pediatrics, in hot water, eating, and somatosen- reflex seizures induced by mic- Nagano Prefectural Suzaka Hospital, 1332 Suzaka, Suzaka City, 382-0091, Japan sory stimulation can also induce turition and/or defecation is also <[email protected]> seizures (Ritaccio, 1994). Micturition reviewed. 166 Epileptic Disord, Vol. 13, No. 2, June 2011 Excretion-induced reflex seizures Case report increased blood flow in her left angular gyrus and left temporal lobe, and decreased flow in her left hip- A five-year-old girl had a mild delay in language deve- pocampus. An interictal electroencephalogram (EEG) lopment and received speech therapy. She had no showed continuous 20-Hz beta activity in the frontal history of febrile convulsions or head injury. Her uncle region, intermittent high-voltage theta waves in the had epilepsy, but detailed information could not be frontal and central regions, and some spike-and-waves obtained. in Cz (figure 1). An ictal EEG recorded on micturition At the age of six years, she was admitted to a nearby revealed the initiation of rhythmic theta waves through hospital because of recurrent generalised tonic-clonic the central electrodes (figure 2). seizures (GTCS) without fever. Several days later, she She was diagnosed with focal epilepsy with reflex developed another type of seizure immediately fol- seizures on the basis of her seizure manifestations lowing micturition, characterised by brief growling, and ictal EEG findings. Her clinical course is shown in eye-opening, the symmetrical extension of both arms figure 3 . The administration of carbamazepine (CBZ) as if to hug or hold someone, and rhythmic jerking resulted in significant decreases in the frequency of of her upper body. These seizures lasted for 10 to 20 both GTCS and reflex seizures, but was discontinued seconds. She remained conscious and reported expe- because of an allergic rash. Zonisamide was not bene- riencing fear at the time of these seizures, and thus ficial and did not reduce the frequency of seizures. The became hesitant to urinate. Since both types of seizure patient then developed brief tonic partial seizures of occurred one to two times daily even after the admi- her left shoulder which occurred in clusters, 39 days nistration of sodium valproate (up to 30 mg/kg/day), after the onset of her seizures. Her reflex seizures she was transferred to our hospital, 14 days after the began to occur not only upon every micturition but onset of her seizures. also on defecation. Clobazam (CLB) was started on On admission, she was alert and physical, neurological, day 41 after seizure onset, at gradually increasing blood, and cerebrospinal fluid examinations revealed doses. Frequent seizures prompted us to begin con- no abnormalities. The KIDS questionnaire-based tinuous intravenous infusion of midazolam (MDZ) on developmental scale evaluated her developmental day 43. However, the medication made her somewhat quotient (DQ) as 65. No abnormalities were detected drowsy and her seizure frequency increased to more on brain magnetic resonance imaging (MRI). Interic- than 60 per day. Treatment with phenytoin (PHT) was th tal 99mTc-ethyl-cysteinate dimer (ECD) single photon attempted on the 46 day in progressively increasing emission computed tomography (SPECT) indicated doses. Despite PHT levels higher than 40 ␮g/mL, the Fp1-A1 Fp2-A2 F3-A1 F4-A2 C3-A1 C4-A2 P3-A1 P4-A2 O1-A1 O2-A2 F7-A1 F8-A2 T3-A1 T4-A2 T5-A1 T6-A2 Fz-A1 Cz-A2 Cz-A1 E-X9 Resp PG1-PG2 ECG Figure 1. Interictal electroencephalography recorded during sleep. Epileptic Disord, Vol. 13, No. 2, June 2011 167 T. Higuchi, et al. Micturition started Seizure started Seizure ended x1 1 Fp1-A1 2 Fp2-A2 3 F3-A1 4 F4-A2 5 C3-A1 6 C4-A2 7 P3-A1 8 P4-A2 9 O1-A1 10 O2-A2 11 F7-A1 12 F8-A2 13 T3-A1 14 T4-A2 15 T5-A1 16 T6-A2 17 Fz-A1 18 Cz-A2 19 Cz-A1 20 E-X9 21 PG1-PG2 M Figure 2. Ictal electroencephalography recorded on micturition. VPA MDZ CBZ ZNS PHT CLB CLB DMCLB (µg/mL) (µg/mL) (ng/mL) PHT 50 500 5,000 40 DMCLB 400 4,000 30 300 3,000 PHT Serum 20 200 2,000 concentration 10 100 1,000 CLB 0 0 0 60 40 (times/day) 20 Seizure frequency 0 13 20 30 40 50 60 Days after onset Figure 3. Clinical and laboratory course. CBZ: carbamazepine; CLB: clobazam; DMCLB: desmethylclobazam; MDZ: midazolam; PHT: phenytoin; VPA: sodium valproate; ZNS: zonisamide. Serum concentrations of PHT, CLB, and DMCLB are shown. 168 Epileptic Disord, Vol. 13, No. 2, June 2011 Excretion-induced reflex seizures seizures did not decrease in frequency and so the dose only at Cz on interictal EEG records and seizure acti- was lowered. The seizures disappeared on the 54th vity started at Cz. The electrical activity in the SMA is day after seizure onset, as the serum concentration recorded at or near the Cz electrode on EEG. In six of of desmethylclobazam (DMCLB), the major metabo- seven cases with evident EEG foci, the seizure focus lite of CLB which also exhibits antiepileptic properties, was observed at Cz, C3, or F4 (table 1). This supports increased. She subsequently had no seizures for two the possibility that the SMA is a seizure focus in many years with the combination of 11 mg/day of CLB and cases of reflex seizures induced by micturition and/or 120 mg/day of PHT. The serum concentrations of CLB, defecation. DMCLB, and PHT were 110-180 (reference range: 50- The reflex seizures induced by micturition or defe- 300) ␮g/mL, 1,400-2,200 (reference range: 1,000-4,000) cation in this case were characterised by tonic ng/mL and 9.7-12.1 (reference range: 10-20) ␮g/mL, posturing of the upper limbs, preserved conscious- respectively. When the PHT dose was decreased in ness, and growling, and suggested involvement of a stepwise fashion to 50 mg/day, micturition-induced a common neuronal pathway. These characteristics seizures occurred three times in one week. At that closely resembled those of reflex seizures in cases time, serum concentrations of CLB, DMCLB, and reported previously (Ikeno et al., 1998; Glass et al., PHT were 132 ␮g/mL, 1,442 ng/mL, and 3.1 ␮g/mL, 2006; Okumura et al., 2007). Bass et al. clarified, using respectively. Accordingly, the PHT dose was raised to video-EEG and subdural electrodes, that such seizure 110 mg/day, which resulted in complete control of the attacks, characterised by sudden tonic posturing of the seizures. limbs and often accompanied by unintelligible fearful vocalization, were derived from the SMA (Bass et al., 1995). These findings also provide supporting evidence Discussion that seizure activity induced by micturition/defecation originates in the SMA in some cases. Reflex seizures often occur together with unprovoked Reflex seizures generally respond to conventional seizures and are rarely triggered by micturition and antiepileptic drugs (Ferlazzo et al., 2005). However, defecation. In the literature, we were able to find only there is no standard antiepileptic drug regime effec- nine reports of reflex seizures induced by micturition tive against all reflex seizures, as illustrated in nine or defecation, but not by both, (Zivin and Rowley, 1964; reported cases of patients who achieved remission Yamatani et al., 1987; Pradhan and Kalita, 1993; Ikeno et with different drugs (table 1). In our case, her seizures al., 1998; Bourgeois, 1999; Harbord and Mitchell, 2002; responded well to CBZ at the beginning of the clinical Glass et al., 2006; Okumura et al., 2007) (table 1). Six course. After the discontinuation of CBZ, because of cases were also reported with unprovoked seizures, a skin rash, her seizures became uncontrolled.
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