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An autopsy case of bathtub drowning in epilepsy
Brain & Development 19 (1997) 499–501
Case report
An autopsy case of bathtub drowning in epilepsy Toshio Osamura a ,*, Shinji Fushiki b, Hiroshi Yoshioka c, Tatsuhiro Yamanaka d, Ryuzo Mizuta a a
Department of Pediatrics, Kyoto Second Red Cross Hospital, Marutamachi, Kamanzadori, Kamigyo-ku, Kyoto 602, Japan b Department of Dynamic Pathology, Kyoto Prefectural University of Medicine, Kyoto, Japan c Department of Pediatrics, Kyoto Prefectural University of Medicine, Kyoto, Japan d Department of Child Health, National Children’s Castle, Tokyo, Japan Received 7 April 1997; revised version received 22 July 1997; accepted 28 July 1997
Abstract We report an autopsy case of bathtub drowning in epilepsy. A 26-year-old female with mental retardation had been treated for refractory epilepsy. Her younger sister found her floating supine in the bathtub 45 min after starting bathing. Neuropathological examination revealed cerebral cortical dysplasia in the precentral gyrus of the left hemisphere, which had not been detected by MRI, suggesting the etiology of epilepsy. In bathtub submersion injury of an unidentified cause, neuropathological examination should be performed to reveal any lesion underlying epileptic seizures. Additionally, we present statistics on bathtub submersion injury in children aged 5 years or older in Japan based upon nationwide survey data obtained in 1991. Forty-seven percent of them had associated epilepsy or convulsive attacks and 71% died. It is necessary for epileptic patients and their families to understand that the risk of bathtub drowning can be minimized if proper precautions are taken. 1997 Elsevier Science B.V. Keywords: Bathtub drowning; Epilepsy; Cortical dysplasia
1. Introduction Previous studies on submersion injury in children [1–4] have disclosed the following: (i) the incidence of submersion injury associated with epileptic seizures is 4–6% of all the submersion injuries; (ii) the incidence of submersion injury in children with epilepsy is 4–10 times that in children without epilepsy; (iii) the mortality rate while swimming with supervision is relatively low, although epileptic seizures may occur while swimming; and (iv) most cases of drowning of children with epilepsy occur in the bathtub at home without any witnesses. According to Diekema et al. [4], the incidence of submersion injury in children with epilepsy is significantly higher when they are aged 5 years or older. In Japan, where bathing daily in hot water is common, the incidence of submersion injury in the bathtub is 5– 6 times [5] that in the United States. Therefore, instructions for daily living for older children with epilepsy should
* Corresponding author. Fax: +81 75 256 3451.
0387-7604/97/$17.00 1997 Elsevier Science B.V. All rights reserved PII S0387-7604 (97 )0 0064-8
include strategies for preventing submersion injury in the bathtub, especially in Japan. We report an autopsy case of bathtub drowning who had been treated for refractory epilepsy with mental retardation (MR). Additionally, in this report, we present statistics on bathtub submersion injury in children aged 5 years or older in Japan based upon nationwide survey data on submersion injury in children obtained in 1991 [5], and discuss the high risk of bathtub drowning in older children with epilepsy as well as the necessity of neuropathological examination of drowned patients.
2. Case report 2.1. Patient The patient (a 26-year-old female) was delivered at 36 weeks of gestation (birth weight, 2200 g) without asphyxia. Mild paralysis of the left lower limb and mild MR (IQ: 50– 60) were noted, although assistance in daily living was
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T. Osamura et al. / Brain & Development 19 (1997) 499–501
unnecessary. Under the diagnosis of symptomatic localization-related epilepsy, she had received treatment since 5 years of age. However, a right-side tonic seizure or generalized tonic-clonic seizure occurred once every 2 or 3 months, and complex partial seizures with automatism of the mouth and fingers occurred several times a month. EEG showed slow wave dysrhythmia in background activity and sometimes sporadic spikes in the left parietal lobe. Magnetic resonance imaging (MRI) of the head did not show any abnormality suggestive of focus of epilepsy. She took a bath by herself from 1800 h, but had not come out of the bathroom by 1845 h. Her younger sister went to the bathroom and found her floating supine in the bathtub with cardiopulmonary arrest. There was no evidence of splashing or struggle in the bathroom. She was brought to the Kyoto Second Red Cross Hospital but was pronounced dead on arrival at 1930 h. She used to bathe everyday by herself, because her parents had not experienced her having a seizure in the bathroom. Furthermore, electrocardiography performed at an institution for handicapped children had revealed no abnormality. There was no tendency toward low compliance regarding medication with antiepileptic drugs (AED). The blood concentrations of AED (phenytoin and carbamazepine) measured at our outpatient clinic 1 month prior to the drowning were within the therapeutic ranges. Although cranial CT was performed, with the consent of her family, no high-density lesion suggesting hemorrhage was detected in the brain. 2.2. Autopsy findings Autopsy was performed 16 h after death. There was no trauma or congestive patches on the surface skin. Obstruction associated with foreign substances or secretion was not observed in the respiratory tract. There was no swallowed water in the stomach. In the lungs, marked congestion and
hemorrhage were noted. In the heart, there were no abnormal findings. The brain weight was 1284 g. Microscopically, in the left precentral gyrus, the cortical architecture was partially disorganized with disturbed polarity in some of the neurons (Fig. 1A). In addition, there were scattered neurons in the cerebral white matter, suggesting microscopic heterotopia (Fig. 1B). A neuropathological diagnosis of cerebral cortical dysplasia was made based on the abovementioned findings. The number of pyramidal cells of Ammon’s horn in the hippocampus was decreased.
3. Discussion In general, there is no difference in autopsy findings in the respiratory tract and heart between submersion from epileptic seizures and other drowning. Autopsy in our case revealed marked congestion and hemorrhage in the lungs, which were consistent with the initial stage of shock lung. Shock lung is a pathological change often seen in drowning. No additional changes which may result in death were noted in the respiratory tract or in the heart. Neuropathological examination in the patient showed cortical dysplasia with a disorganized cortical architecture and microscopic heterotopias involving the left precentral gyrus. Cortical dysplasia is considered to be a neuronal migration disorder, which is closely associated with epilepsy and/or MR [6]. Although she had mild MR, the cortical dysplasia found in the left precentral gyrus may well correspond to the focus of right hemiconvulsions as well as the spike site on EEG. Therefore, in this patient, the drowning may have been associated with a convulsive attack during bathing. Desbiens et al. [6] reported that seizures due to focal cortical dysplasia with normal MRI are often intractable and might be life-threatening. In 1991, Mizuta conducted a nationwide survey concern-
Fig. 1. Histology of the left precentral gyrus (hematoxylin and eosin staining). (A) Abnormality of the cortical lamination with disturbed polarity of neurons can be seen in the deep cortex. Scale bar, 100 mm. (B) Neurons are scattered in the white matter. Arrowheads indicate ectopic neurons. Scale bar, 100 mm.
501
T. Osamura et al. / Brain & Development 19 (1997) 499–501 Table 1 Bathtub submersion at home in children aged 5 years or older in Japan (based on a national survey conducted by means of a questionnaire in 1991) No.
Age (years)
Gender
Duration of submersion
Prognosis
Past history
Situation of submersion
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15
5 5 5 5 5 6 6 7 8 11 12 12 12 17 18
Girl Boy Boy Girl Girl Boy Boy Girl Boy Boy Boy Girl Girl Girl Boy
1–2 min 2–3 min Several min Several min 30 min 1–2 min 4–5 min 5–10 min Unknown 5 min More than 30 min Unknown Unknown 20–30 min Unknown
Good Good Good Good Death Good Good Good Good Good Death Death Death Death Death
– – – Febrile convulsion – Epilepsy – – Mental retardation – – Epilepsy – Epilepsy Epilepsy
Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Convulsion occurred during bathing Convulsion occurred during bathing Low compliance with medication Convulsion occurred during bathing Low compliance with medication Unremarkable
ing submersion injury in children in Japan [5]. The survey showed that 604 children aged 0–18 years had experienced submersion between 1974 and 1991. Of the 604, 15 (11%) were 5 years or older and had experienced submersion injury in the bathtub at home. The situations during submersion are shown in Table 1. Seven of the 15 children (47%) were patients with a past history of epilepsy or patients in whom a convulsive attack during bathing may have resulted in the submersion injury. Five of them (71%) died. This suggests that bathtub submersion in older children may be closely associated with epilepsy. Many researchers have indicated that the high mortality due to bathtub submersion in older children is associated with their habit of bathing by themselves and delayed notice of the submersion injury [4,7,8]. Therefore, pediatricians and pediatric neurologists should inform patients with epilepsy as well as their families of the risk involved in bathing alone. Special precautions should be taken to prevent submersion injury, when patients have both epilepsy and MR, because the risk of submersion injury is increased among them [1–3]. It is not always possible to supervise older children while bathing. However, the risk of bathtub death may be minimized by making their families aware that they are bathing whenever they do, leaving the bathroom door unlocked, and using a hand shower in the sitting position in the bathtub without a bathtub stopper [4,8,9]. In addition, it should be noted that some epileptic seizures are easily induced by hot bathing. Therefore, patients suffering from severe myoclonic epilepsy in infants or symptomatic epilepsy with febrile convulsions and with seizures induced by hot bathing should take a shower with tepid water. Furthermore, a convulsive attack while bathing due to low medication compliance may have been the cause of death in Nos. 12 and 14 in Table 1. The need for regular medication should be emphasized in patients showing low compliance with AED. Bathtub submersion, differing from drowning in the river or sea, can be avoided by adequate precautions, and the instructions as to daily living given above are very important.
In Nos. 11 and 13 in Table 1, the drowning may have been associated with an initial epileptic seizure. It is known that bathtub submersion is sometimes associated with an initial epileptic seizure [3,10]. Therefore, in bathtub submersion injury, especially in older children, even though the patients do not have a previous history of a convulsive disease, neuropathological examination should be performed, because autopsy may disclose underlying cerebral lesions associated with epilepsy.
Acknowledgements This study was supported in part by grants from the Ministry of Health and Welfare of Japan. References [1] Pearn JH. Epilepsy and drowning in childhood. Br Med J 1977;11:1510–1511. [2] Orlowski JP, Rothner AD, Lueders H. Submersion accidents in children with epilepsy. Am J Dis Child 1982;136:777–780. [3] Kemp AM, Sibert JR. Epilepsy in children and the risk of drowning. Arch Dis Child 1993;68:684–685. [4] Diekema DS, Quan L, Holt VL. Epilepsy as a risk factor for submersion injury in children. Pediatrics 1993;91:612–616. [5] Mizuta R, Fujita H, Osamura T, Kidowaki T, Kiyosawa N. Childhood drownings and near-drownings in Japan. Acta Paediatr Jpn 1993;35:186–192. [6] Desbiens R, Berkovic SF, Dubeau F, Andermann F, Laxen KD, Harvey S. et al. Life-threatening focal status epilepticus due to occult cortical dysplasia. Arch Neurol 1993;50:695–700. [7] Yagi K, Suzuki S, Seino M. Unexpected death in 55 patients with epilepsy. Folia Psychiat Neurol Jpn 1985;39:391–392. [8] Ryan CA, Dowling G. Drowning deaths in people with epilepsy. Can Med Assoc J 1993;148:781–784. [9] Livingston S, Pauli LL, Kramer IP, Kramer II. Drowning in epilepsy. Ann Neurol 1980;7:495. [10] Saxena A, Ang LC. Epilepsy and bathtub drowning, important neuropathological observations. Am J Forensic Med Pathol 1993; 14:125–129.
Case report
An autopsy case of bathtub drowning in epilepsy Toshio Osamura a ,*, Shinji Fushiki b, Hiroshi Yoshioka c, Tatsuhiro Yamanaka d, Ryuzo Mizuta a a
Department of Pediatrics, Kyoto Second Red Cross Hospital, Marutamachi, Kamanzadori, Kamigyo-ku, Kyoto 602, Japan b Department of Dynamic Pathology, Kyoto Prefectural University of Medicine, Kyoto, Japan c Department of Pediatrics, Kyoto Prefectural University of Medicine, Kyoto, Japan d Department of Child Health, National Children’s Castle, Tokyo, Japan Received 7 April 1997; revised version received 22 July 1997; accepted 28 July 1997
Abstract We report an autopsy case of bathtub drowning in epilepsy. A 26-year-old female with mental retardation had been treated for refractory epilepsy. Her younger sister found her floating supine in the bathtub 45 min after starting bathing. Neuropathological examination revealed cerebral cortical dysplasia in the precentral gyrus of the left hemisphere, which had not been detected by MRI, suggesting the etiology of epilepsy. In bathtub submersion injury of an unidentified cause, neuropathological examination should be performed to reveal any lesion underlying epileptic seizures. Additionally, we present statistics on bathtub submersion injury in children aged 5 years or older in Japan based upon nationwide survey data obtained in 1991. Forty-seven percent of them had associated epilepsy or convulsive attacks and 71% died. It is necessary for epileptic patients and their families to understand that the risk of bathtub drowning can be minimized if proper precautions are taken. 1997 Elsevier Science B.V. Keywords: Bathtub drowning; Epilepsy; Cortical dysplasia
1. Introduction Previous studies on submersion injury in children [1–4] have disclosed the following: (i) the incidence of submersion injury associated with epileptic seizures is 4–6% of all the submersion injuries; (ii) the incidence of submersion injury in children with epilepsy is 4–10 times that in children without epilepsy; (iii) the mortality rate while swimming with supervision is relatively low, although epileptic seizures may occur while swimming; and (iv) most cases of drowning of children with epilepsy occur in the bathtub at home without any witnesses. According to Diekema et al. [4], the incidence of submersion injury in children with epilepsy is significantly higher when they are aged 5 years or older. In Japan, where bathing daily in hot water is common, the incidence of submersion injury in the bathtub is 5– 6 times [5] that in the United States. Therefore, instructions for daily living for older children with epilepsy should
* Corresponding author. Fax: +81 75 256 3451.
0387-7604/97/$17.00 1997 Elsevier Science B.V. All rights reserved PII S0387-7604 (97 )0 0064-8
include strategies for preventing submersion injury in the bathtub, especially in Japan. We report an autopsy case of bathtub drowning who had been treated for refractory epilepsy with mental retardation (MR). Additionally, in this report, we present statistics on bathtub submersion injury in children aged 5 years or older in Japan based upon nationwide survey data on submersion injury in children obtained in 1991 [5], and discuss the high risk of bathtub drowning in older children with epilepsy as well as the necessity of neuropathological examination of drowned patients.
2. Case report 2.1. Patient The patient (a 26-year-old female) was delivered at 36 weeks of gestation (birth weight, 2200 g) without asphyxia. Mild paralysis of the left lower limb and mild MR (IQ: 50– 60) were noted, although assistance in daily living was
500
T. Osamura et al. / Brain & Development 19 (1997) 499–501
unnecessary. Under the diagnosis of symptomatic localization-related epilepsy, she had received treatment since 5 years of age. However, a right-side tonic seizure or generalized tonic-clonic seizure occurred once every 2 or 3 months, and complex partial seizures with automatism of the mouth and fingers occurred several times a month. EEG showed slow wave dysrhythmia in background activity and sometimes sporadic spikes in the left parietal lobe. Magnetic resonance imaging (MRI) of the head did not show any abnormality suggestive of focus of epilepsy. She took a bath by herself from 1800 h, but had not come out of the bathroom by 1845 h. Her younger sister went to the bathroom and found her floating supine in the bathtub with cardiopulmonary arrest. There was no evidence of splashing or struggle in the bathroom. She was brought to the Kyoto Second Red Cross Hospital but was pronounced dead on arrival at 1930 h. She used to bathe everyday by herself, because her parents had not experienced her having a seizure in the bathroom. Furthermore, electrocardiography performed at an institution for handicapped children had revealed no abnormality. There was no tendency toward low compliance regarding medication with antiepileptic drugs (AED). The blood concentrations of AED (phenytoin and carbamazepine) measured at our outpatient clinic 1 month prior to the drowning were within the therapeutic ranges. Although cranial CT was performed, with the consent of her family, no high-density lesion suggesting hemorrhage was detected in the brain. 2.2. Autopsy findings Autopsy was performed 16 h after death. There was no trauma or congestive patches on the surface skin. Obstruction associated with foreign substances or secretion was not observed in the respiratory tract. There was no swallowed water in the stomach. In the lungs, marked congestion and
hemorrhage were noted. In the heart, there were no abnormal findings. The brain weight was 1284 g. Microscopically, in the left precentral gyrus, the cortical architecture was partially disorganized with disturbed polarity in some of the neurons (Fig. 1A). In addition, there were scattered neurons in the cerebral white matter, suggesting microscopic heterotopia (Fig. 1B). A neuropathological diagnosis of cerebral cortical dysplasia was made based on the abovementioned findings. The number of pyramidal cells of Ammon’s horn in the hippocampus was decreased.
3. Discussion In general, there is no difference in autopsy findings in the respiratory tract and heart between submersion from epileptic seizures and other drowning. Autopsy in our case revealed marked congestion and hemorrhage in the lungs, which were consistent with the initial stage of shock lung. Shock lung is a pathological change often seen in drowning. No additional changes which may result in death were noted in the respiratory tract or in the heart. Neuropathological examination in the patient showed cortical dysplasia with a disorganized cortical architecture and microscopic heterotopias involving the left precentral gyrus. Cortical dysplasia is considered to be a neuronal migration disorder, which is closely associated with epilepsy and/or MR [6]. Although she had mild MR, the cortical dysplasia found in the left precentral gyrus may well correspond to the focus of right hemiconvulsions as well as the spike site on EEG. Therefore, in this patient, the drowning may have been associated with a convulsive attack during bathing. Desbiens et al. [6] reported that seizures due to focal cortical dysplasia with normal MRI are often intractable and might be life-threatening. In 1991, Mizuta conducted a nationwide survey concern-
Fig. 1. Histology of the left precentral gyrus (hematoxylin and eosin staining). (A) Abnormality of the cortical lamination with disturbed polarity of neurons can be seen in the deep cortex. Scale bar, 100 mm. (B) Neurons are scattered in the white matter. Arrowheads indicate ectopic neurons. Scale bar, 100 mm.
501
T. Osamura et al. / Brain & Development 19 (1997) 499–501 Table 1 Bathtub submersion at home in children aged 5 years or older in Japan (based on a national survey conducted by means of a questionnaire in 1991) No.
Age (years)
Gender
Duration of submersion
Prognosis
Past history
Situation of submersion
1 2 3 4 5 6 7 8 9 10 11 12 13 14 15
5 5 5 5 5 6 6 7 8 11 12 12 12 17 18
Girl Boy Boy Girl Girl Boy Boy Girl Boy Boy Boy Girl Girl Girl Boy
1–2 min 2–3 min Several min Several min 30 min 1–2 min 4–5 min 5–10 min Unknown 5 min More than 30 min Unknown Unknown 20–30 min Unknown
Good Good Good Good Death Good Good Good Good Good Death Death Death Death Death
– – – Febrile convulsion – Epilepsy – – Mental retardation – – Epilepsy – Epilepsy Epilepsy
Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Unremarkable Convulsion occurred during bathing Convulsion occurred during bathing Low compliance with medication Convulsion occurred during bathing Low compliance with medication Unremarkable
ing submersion injury in children in Japan [5]. The survey showed that 604 children aged 0–18 years had experienced submersion between 1974 and 1991. Of the 604, 15 (11%) were 5 years or older and had experienced submersion injury in the bathtub at home. The situations during submersion are shown in Table 1. Seven of the 15 children (47%) were patients with a past history of epilepsy or patients in whom a convulsive attack during bathing may have resulted in the submersion injury. Five of them (71%) died. This suggests that bathtub submersion in older children may be closely associated with epilepsy. Many researchers have indicated that the high mortality due to bathtub submersion in older children is associated with their habit of bathing by themselves and delayed notice of the submersion injury [4,7,8]. Therefore, pediatricians and pediatric neurologists should inform patients with epilepsy as well as their families of the risk involved in bathing alone. Special precautions should be taken to prevent submersion injury, when patients have both epilepsy and MR, because the risk of submersion injury is increased among them [1–3]. It is not always possible to supervise older children while bathing. However, the risk of bathtub death may be minimized by making their families aware that they are bathing whenever they do, leaving the bathroom door unlocked, and using a hand shower in the sitting position in the bathtub without a bathtub stopper [4,8,9]. In addition, it should be noted that some epileptic seizures are easily induced by hot bathing. Therefore, patients suffering from severe myoclonic epilepsy in infants or symptomatic epilepsy with febrile convulsions and with seizures induced by hot bathing should take a shower with tepid water. Furthermore, a convulsive attack while bathing due to low medication compliance may have been the cause of death in Nos. 12 and 14 in Table 1. The need for regular medication should be emphasized in patients showing low compliance with AED. Bathtub submersion, differing from drowning in the river or sea, can be avoided by adequate precautions, and the instructions as to daily living given above are very important.
In Nos. 11 and 13 in Table 1, the drowning may have been associated with an initial epileptic seizure. It is known that bathtub submersion is sometimes associated with an initial epileptic seizure [3,10]. Therefore, in bathtub submersion injury, especially in older children, even though the patients do not have a previous history of a convulsive disease, neuropathological examination should be performed, because autopsy may disclose underlying cerebral lesions associated with epilepsy.
Acknowledgements This study was supported in part by grants from the Ministry of Health and Welfare of Japan. References [1] Pearn JH. Epilepsy and drowning in childhood. Br Med J 1977;11:1510–1511. [2] Orlowski JP, Rothner AD, Lueders H. Submersion accidents in children with epilepsy. Am J Dis Child 1982;136:777–780. [3] Kemp AM, Sibert JR. Epilepsy in children and the risk of drowning. Arch Dis Child 1993;68:684–685. [4] Diekema DS, Quan L, Holt VL. Epilepsy as a risk factor for submersion injury in children. Pediatrics 1993;91:612–616. [5] Mizuta R, Fujita H, Osamura T, Kidowaki T, Kiyosawa N. Childhood drownings and near-drownings in Japan. Acta Paediatr Jpn 1993;35:186–192. [6] Desbiens R, Berkovic SF, Dubeau F, Andermann F, Laxen KD, Harvey S. et al. Life-threatening focal status epilepticus due to occult cortical dysplasia. Arch Neurol 1993;50:695–700. [7] Yagi K, Suzuki S, Seino M. Unexpected death in 55 patients with epilepsy. Folia Psychiat Neurol Jpn 1985;39:391–392. [8] Ryan CA, Dowling G. Drowning deaths in people with epilepsy. Can Med Assoc J 1993;148:781–784. [9] Livingston S, Pauli LL, Kramer IP, Kramer II. Drowning in epilepsy. Ann Neurol 1980;7:495. [10] Saxena A, Ang LC. Epilepsy and bathtub drowning, important neuropathological observations. Am J Forensic Med Pathol 1993; 14:125–129.