Ictal EEG in patients with convulsions with mild gastroenteritis

Ictal EEG in patients with convulsions with mild gastroenteritis

Brain & Development 29 (2007) 43–46 www.elsevier.com/locate/braindev Ictal EEG in patients with convulsions with mild gastroenteritis Koichi Maruyama...

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Brain & Development 29 (2007) 43–46 www.elsevier.com/locate/braindev

Ictal EEG in patients with convulsions with mild gastroenteritis Koichi Maruyama a,b,*, Akihisa Okumura a,c, Ayako Sofue b, Naoko Ishihara d, Kazuyoshi Watanabe e a

Department of Pediatrics, Anjo Kosei Hospital, 28 Higashi-Hirokute, Anjo-cho, Anjo, Aichi 446-8602, Japan b Department of Pediatrics, Japan Red Cross Nagoya First Hospital, Nagoya, Japan c Department of Pediatrics, Nagoya University Graduate School of Medicine, Nagoya, Japan d Department of Pediatrics, Kamo Hospital, Toyota, Japan e Faculty of Medical Welfare, Aichi Shukutoku University, Nagoya, Japan Received 16 March 2006; received in revised form 10 June 2006; accepted 13 June 2006

Abstract The aim of this study is to reveal detailed clinical manifestations and an evolution of ictal EEG discharges of convulsions with mild gastroenteritis (CwG). We recorded ictal EEGs of six patients with CwG. Clinical manifestations included loss of responsiveness, motion arrest, cyanosis, lateral eye deviation, and hemifacial convulsion. Automatism was not observed in any patients. A generalized tonic–clonic convulsion was observed in five of six patients. Ictal EEGs demonstrated that all seizures were of focal onset that evolved into a secondarily generalized seizure. The region of the onset of ictal discharge was the occipital area in three patients, parietal in one, central in one, and frontal in one, respectively. The seizure of patients with CwG is likely to be a partial seizure with secondary generalization.  2006 Elsevier B.V. All rights reserved. Keywords: Convulsion with mild gastroenteritis; Benign partial epilepsy in infancy; Ictal EEG

1. Introduction In 1982, Morooka first reported benign convulsions with mild gastroenteritis [1]. Since then, clinical features of convulsions with mild gastroenteritis (CwG) have been described in several reports [2–4]. At present, CwG is popular and well recognized among pediatricians in Japan. CwG is characterized by: (a) afebrile generalized seizures associated with symptoms of gastroenteritis in previously healthy infants or young children aged between six months and three years; (b) seizures frequently occurring in clusters; (c) normal laboratory examination *

Corresponding author. Tel.: +81 566 75 2111; fax: +81 566 76 4335. E-mail address: [email protected] (K. Maruyama). 0387-7604/$ - see front matter  2006 Elsevier B.V. All rights reserved. doi:10.1016/j.braindev.2006.06.002

results including serum electrolytes, glucose, and cerebrospinal fluid; (d) normal interictal electroencephalogram (EEG); and (e) excellent seizure and developmental outcome [1–4]. The seizures of patients with CwG have been often described as symmetrical generalized convulsions. However, some of the seizures had some focal features such as lateral eye deviation and hemiconvulsion [2] or resembled complex partial ones such as loss of responsiveness without convulsive movements [4]. Reports of ictal EEGs were rare, but they demonstrated that the seizures of patients with CwG were secondarily generalized ones [5–8]. In the present study, we recorded ictal EEGs of six patients in order to clarify detailed clinical manifestations and an evolution of EEG discharges in patients with CwG.

89 12 PS-SGS Bilateral frontal pole 6 Positive – – F 6

3:2

82 9 PS-SGS Left central 6 Negative Fc CwG F 5

2:7

85 6 PS-SGS Bilateral occipital 6 Negative – CwG F 4

2:7

91 12 PS-SGS Left parietal 4 Positive – – M 3

2:3

– F 2

2:1

M 1

CwG, convulsion with gastroenteritis; Fc, febrile convulsion; NA, not available; ND, not detected; PS, partial seizure; SGS, secondarily generalized seizure; GTCS, generalized tonic–clonic seizure.

1:5

3:3

5:2

2:11

2:9 Negative

4

Left occipital

PS-SGS

20

125

Loss of responsiveness, right lateral gaze, motion arrest and cyanosis fi GTCS Loss of responsiveness with vocalization, blank eyes and cyanosis fi GTCS Loss of responsiveness, motion arrest fi GTCS Loss of responsiveness, left lateral gaze, left hemifacial convulsion fi GTCS Loss of responsiveness, upward gaze fi left lateral gaze with cyanosis fi GTCS Loss of responsiveness fi right hemiconvulsion 93 9 PS-SGS Right occipital 4 ND

CwG, infantile seizure NA

Origin of ictal discharge No. of seizure in cluster Rotavirus antigen in stool Family history Past history Age (years: months) Sex

Table 1 Profiles of 6 patients with convulsion with gastroenteritis

The profiles of patients were shown in Table 1. There were two boys and four girls. The age of onset ranged from 14 months to 38 months (mean, 27.7 ± 8.0 months). A past history of CwG was noted in two patients (patients 4 and 5), but that of febrile convulsion was not present. A familial case of CwG (patient 1) was a pair of identical twins. Their clinical details were documented elsewhere [6]. A family history for CwG was positive in patients 1 and 5. The mother and aunt of patient 1 had histories of afebrile seizures during infancy. The co-twin of patient 5 had a history of febrile convulsions. The interval between the onset of gastroenteritis and that of convulsions ranged 0–3 days (mean, 2.2 ± 1.3 days). A cluster of seizures was observed in all patients, and the number of seizures ranged 4-6 (mean, 5.0 ± 1.1). Rotavirus antigen test in stool was positive in two of five patients examined. The clinical manifestation was similar to that of a partial seizure. Loss of responsiveness and motion arrest were seen in all patients, cyanosis in four, lateral eye deviation in three (ipsilateral to the origin in two and contralateral to the origin in one), and hemifacial convulsion in one. Oral, facial, or behavioral automatisms were not observed in any patients. A generalized tonic–clonic convulsion was documented in five of six patients (patients 1–5). Ictal EEGs demonstrated paroxysmal discharges beginning as low amplitude fast rhythm recruiting from some limited region in all patients. Representative ictal EEGs were shown in Figs. 1 and 2. The region of onset

Seizure evolution

3. Result



Time to secondary generalization (s)

Duration of seizure (s)

Clinical manifestation

The subjects of this study were six patients with CwG whose ictal EEG was recorded in the Departments of Pediatrics in Anjo Kosei Hospital, Japan Red Cross Nagoya First Hospital, and Kamo Hospital between November 1996 and March 2005. The definition of CwG was the same as that in the previous studies [4,9]. CwG was diagnosed when a patient met both of the following: (a) seizures accompanied gastroenteritis without clinical signs of dehydration or electrolyte imbalance; and (b) a body temperature of less than 38.0 C before and after the seizure. Patients with meningitis, encephalitis/encephalopathy, or an apparent history of epilepsy were excluded. Because simultaneous video-EEG recording was not available in our hospitals, clinical manifestations were judged according to the observations of physicians or technicians. Clinical courses, family and past histories, and prognosis were investigated on the basis of medical records.

1:2

Duration of followup (years: months)

2. Patients and methods

4:0

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Patient no.

44

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Fig. 1. Ictal EEG of patient 2. Seizure discharges originated from left occipital region (A), evolved to secondarily generalized seizure 20 s after the onset (B), and suddenly stopped 125 s after the onset (C).

Fig. 2. Ictal EEG of patient 6. Seizure discharges originated from bilateral frontal pole (A), evolved to secondarily generalized seizure 12 s after the onset (B), and suddenly stopped 89 s after the onset (C).

was the occipital area in three patients, parietal in one, central in one, and frontal in one, respectively. The laterality of the onset region was left in three patients and right in one. In the remaining two patients, seizure discharges occurred bilaterally. The ictal discharge evolved into a secondarily generalized seizure in all patients. The interval from the onset of seizures to generalization ranged 9–20 s (mean, 11.3 ± 4.8 s). The duration of seizures ranged 82–125 s (mean, 94.2 ± 15.6 s). Seizures were controlled with antiepileptic drugs (phenobarbital, lidocaine, or carbamazepine) in all patients. Follow-up periods ranged 17–62 months (mean, 39 ± 16 months). Interictal EEGs were normal, and normal psychomotor development was confirmed at the last follow-up in all patients.

4. Discussion Previous reports documented that most seizures in patients with CwG were symmetrical generalized convulsions [3]. However, some of the seizures were described to have focal features. Uemura et al. [4] analyzed 114 episodes in 105 patients, and reported that 11 patients (13%) had complex partial seizures with loss of responsiveness, eye deviation, cyanosis, and subtle convulsive movements. They did not mention the presence or absence of secondary generalization. Komori et al. [2] analyzed 19 episodes of 10 patients with CwG, and reported that 10 episodes in seven patients had focal features such as hemiconvulsion, lateral gaze, or pedaling-like

movement. The seizures of our patients were accompanied by loss of responsiveness, motion arrest, cyanosis, and focal motor components such as lateral eye deviation and hemiconvulsion. These facts strongly suggest that the seizure in patients with CwG is by its nature a partial seizure with secondary generalization. Our study revealed that the seizure discharges in all patients with CwG originated in a certain limited area and evolved into a secondarily generalized seizure. We found that the site of the origin of seizures varied among patients with CwG. In our patients, three seizures originated in occipital area, whereas the remaining three seizures originated in the parietal, central, and frontal area, respectively. A few authors described ictal video-EEG recordings. They also reported that seizures in patients with CwG were a partial seizure that evolved into a secondarily generalized seizure. Tsurui et al. [5] described the ictal EEG findings of two patients with CwG. One seizure began in the frontal area, and the other from the temporo-parietal area. Capovilla and Vigevano [8] described an ictal EEG in a patient of CwG that originated in left centro-parietal area. These facts indicate that most of seizures in patients with CwG will originate in the centro-parieto-occipital area, a few seizures in frontal area, but none in temporal area. Furthermore, the origin of the seizures in patients with CwG would migrate from one seizure to another even in a cluster of a single patient. In this study, we did not record multiple seizures in a single patient. Imai et al. [7] reported ictal EEGs of three seizures in a single patient. The site of the origin of her seizures migrated among right

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occipital, right centro-parietal, and left occipital area. The symptoms of the seizures were, however, very similar despite the site of origin. It would be difficult to distinguish the site of origin on the basis of clinical manifestation. Although CwG is likely to be categorized as situation-related seizures, it has some features that are similar to those of idiopathic benign partial epilepsies in infancy and early childhood, such as benign partial epilepsy in infancy with secondarily generalized seizures [8,10,11]. They are age-specific, followed by excellent seizure and psychomotor outcome. A cluster of seizures is very common. The distribution of the origin of seizures was closely similar. In some patients, different originating regions were disclosed in a single patient. In the study of 105 patients by Uemura et al. [4], family histories of febrile or afebrile seizures were noted only in 7% and 6% of the patients with CwG, respectively. In our study, however, two of six patients (1 and 5) had a positive family history. Patient 1 and his co-twin were identical to the only cases of familial CwG in the study of Uemura et al. Their mother and aunt also had histories of afebrile seizures during infancy, but the detailed information of the seizures was not available, such as whether gastroenteritis was accompanied or not. We did not perform genetic study, but genetic predisposition might be a contributing factor in genesis of CwG to some extent. In conclusion, the seizure type of CwG is likely to be a partial seizure with secondarily generalization. The origin of the seizure varied among patients from the occipital to the frontal area, but no seizure originated

from the temporal area. Clinical manifestations were similar regardless of the site of origin.

References [1] Morooka K. Convulsions and mild diarrhea (in Japanese). Shonika (Tokyo) 1982;23:131–7. [2] Komori H, Wada M, Eto M, Oki H, Aida K, Fujimoto T. Benign convulsions with mild gastroenteritis: a report of 10 recent cases detailing clinical varieties. Brain Dev 1995;17:334–7. [3] Abe T, Kobayashi M, Araki K, Kodama H, Fujita Y, Shinozaki T, et al. Infantile convulsions with mild gastroenteritis. Brain Dev 2000;22:301–6. [4] Uemura N, Okumura A, Negoro T, Watanabe K. Clinical features of benign convulsions with mild gastroenteritis. Brain Dev 2002;24:745–9. [5] Tsurui S, Oguni H, Fukuyama Y. Analysis of ictal EEG in benign infantile convulsions (in Japanese). Tenkankenkyu 1989;7:160–8. [6] Okumura A, Kato T, Hayakawa F, Kuno K, Watanabe K. Convulsion associated with mild gastroenteritis: occurrence in identical twins on the same day (in Japanese). No To Hattatsu 1999;31:59–62. [7] Imai K, Otani K, Yanagihara K, Li Z, Futagi Y, Ono J, et al. Ictal video-EEG recording of three partial seizures in a patient with benign infantile convulsions associated with mild gastroenteritis. Epilepsia 1999;40:1455–8. [8] Capovilla G, Vigevano F. Benign idiopathic partial epilepsies in infancy. J Child Neurol 2001;16:874–81. [9] Okumura A, Uemura N, Negoro T, Watanabe K. Efficacy of antiepileptic drugs in patients with benign convulsions with mild gastroenteritis. Brain Dev 2004;26:164–7. [10] Watanabe K, Negoro T, Aso K. Benign partial epilepsy with secondarily generalized seizures in infancy. Epilepsia 1993;34:635–8. [11] Watanabe K, Okumura A. Benign partial epilepsies in infancy. Brain Dev 2000;22:296–300.