Clinical commentary with video sequences
Epileptic Disord 2008; 10 (1): 45-8
Hot water epilepsy:
a video case report
of a Caucasian toddler
Berten Ceulemans1,2, Muriel Koninckx3, Kristien Garmyn4,
Marek Wojciechowski3
1
Department of Neurology-Child Neurology, Antwerp University Hospital, Edegem
Epilepsy centre for children and youth, Pulderbos
3
Department of Pediatrics, Antwerp University Hospital, Edegem
4
Department of Pediatrics, HH Hospital, Lier, Belgium
Copyright © 2016 John Libbey Eurotext. Téléchargé par un utilisateur anonyme le 18/05/2016.
2
Received September 1, 2007; Accepted December 26, 2007
ABSTRACT – We present the case report of a 13-month-old Caucasian toddler
with symptoms of loss of consciousness, central cyanosis and uncontrolled
movements of the upper limbs while taking a warm bath. The diagnosis of hot
water epilepsy was supported by an ictal EEG. Hot water epilepsy, also known
as bathing epilepsy or water-immersion epilepsy is, in the Caucasian population, a rare form of benign epilepsy, where seizures are provoked by immersion
in a hot or even just a warm bath. This is the first comprehensive video
publication of a seizure provoked by water-immersion in a Caucasian child.
[Published with video sequences].
doi: 10.1684/epd.2008.0171
Key words: hot water epilepsy, seizures, induced seizures
Correspondence:
B. Ceulemans
Antwerp University Hospital,
Wilrijkstraat 10,
2650 Edegem, Belgium
<[email protected]>
Epileptic Disord Vol. 10, No. 1, March 2008
Hot water epilepsy (HWE), also described as bathing epilepsy or waterimmersion epilepsy, is a benign type
of reflex epilepsy. In this disease, seizures are provoked by an external stimulus consisting of hot or even warm
water (Keipert, 1969, Engel, 2001).
This type of epilepsy has mostly been
described in India, Japan and Turkey,
and is rare in Western countries
(De Keyzer et al. 2005).
Satishchandra has reported a prevalence in South-India as high as 3.6 to
6.9% (Satishchandra et al. 1988,
Grosso et al. 2004). Conversely, the
percentage of patients with HWE
among people with epilepsy is only
0.6% (Bebek et al. 2001).
Most frequently, patients show complex partial seizures with or without
secondary generalisation. Rather exceptional is a primary generalised
tonic-clonic convulsion. Sometimes
seizures are preceded by an aura
consisting of flashbacks, rising epigastric sensations, and staring.
HWE is mostly seen in children (boys
more often than girls), but it has also
been described in adults (Satischandra
et al. 1988, Gururaj et al. 1992, Bebek
et al. 2001, Ioos et al. 2000).
Case report
The patient is a 13-month-old boy
who had been referred to the university hospital for exclusion of Dravet
syndrome. He was referred by the pediatrician after a loss of consciousness
for a few minutes while taking a bath.
Medical history was negative and
clinical examination on admission
was normal. Height and weight were
consistent with age. He had never suffered from febrile seizures and his psychomotor development was normal.
45
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B. Ceulemans, et al.
A
B
C
D
Figure 1. A) 10.45.45; start op seizure; B) 10.45.55; generalisation; C) 10.46.35; during seizure; D) 10.47.15; stop of seizure. Longitudinal
montage, calibration 200 lV, speed 20 sec per page.
Family history revealed the presence of convulsions of
unknown cause in the maternal grandfather and his sister.
Routine blood examination and brain imaging were normal. The postictal EEG revealed no signs of epilepsy or
lateralisation. During hospital admission, a seizure could
be provoked by placing the child in a warm bath
(T = 36.6°C), while taking an EEG (see video sequence).
Twenty seconds later the patient stopped playing, started
to stare and consciousness was altered. He became pale,
had peri-oral cyanosis, became hypotonic and showed
some uncontrolled movements involving both upper
limbs. The nurse took him out of the bath and the seizure
stopped spontaneously after, in total, 1min 30sec. In the
postictal phase, the child fell asleep.
The EEG preceding the seizure showed a polymorphic
curve with a normal background rhythm (figure 1A) for his
age. Stimulation with light flashes did not provoke a
seizure. When the seizure started, there was a paroxysmal
delay of the base rhythm followed by a two to three Hz
pattern with high voltage delta waves of 150-200 lV,
starting in the occipital region and diffusely disseminating
46
over all leads (figure 1A and B). There were no other
epileptic discharges (figure 1C). This episode stopped
shortly after the child was taken out of the bath (figure 1D).
The simultaneously-recorded ECG remained normal.
Avoiding bathing in hot water and just washing the child
with lukewarm (32°C) water seemed to be sufficient to
prevent recurrent seizures. Because the parents were very
anxious, sodium valproate was started. At last follow-up at
the age of three years, all medication was stopped, psychomotor development was normal and the patient was
able to take a warm (37°C) shower without any problem.
During the summer, he could play in an outside pool
without experiencing any convulsions. Mutation analyses
of SCN1A were normal, as expected.
Discussion
HWE, bathing or water immersion epilepsy is a reflex
epilepsy that is frequent in South-India, Turkey and Japan,
but is rather exceptional in Caucasian populations (Lenoir
et al. 1989, Satishchandra et al. 1998, Argumosa et al.
Epileptic Disord Vol. 10, No. 1, March 2008
Copyright © 2016 John Libbey Eurotext. Téléchargé par un utilisateur anonyme le 18/05/2016.
Hot water epilepsy
2002, Incecik et al. 2004, De Keyzer et al. 2005). Seizures
are provoked by contact with hot water (Mouterde et al.
1997, Ioos et al. 2000, Sharma et al. 2002, Incecik et al.
2004). The differences seen between countries can probably be explained by local climate, bathing habits and
genetic factors.
In most European countries, “hot water” means warm
water with a temperature around 37°C, which is the
normal temperature of a bath for infants (Grosso et al.
2004, De Keyser et al. 2005). In Turkey however, a “hot
bath” means water with a temperature of up to 50°C
(Incecik et al. 2004). It has also to be noted that most
Caucasian patients are infants or toddlers, while in the
series from India and Turkey most patients are much older
or even adults.
Although the diagnosis is mostly clinical, EEG has an
important contribution (Satishchandra et al. 1998, Bebek
et al. 2001, Satishchandra, 2003).
Interictal EEG is usually normal, although lateralised or
localised spikes in the anterior temporal region have been
reported in a few isolated cases (Satishchandra, 2003).
An ictal EEG is, for obvious reasons, difficult to obtain. In
those cases where it has been observed, seizures are
accompanied by lateralised rhythmic delta-activity, sharp
and slow waves, sometimes bilateral spikes or localised
temporal activity (Ioos et al. 2000, DiMario, 2001). Pictures of an ictal video-EEG recording of a single patient
were first published by Roos (Roos et al. 1988). In two of
the patients reported by Ioos, diagnosis of HWE was
supported by ictal video-EEG recordings (Ioos et al. 2000).
Here we present, for the first time, a video of a complete
seizure provoked by water immersion in a Caucasian
toddler with HWE.
Differential diagnoses of vagal syncope, breath-holding
spells and febrile seizures can nearly always be made by
careful history-taking (Fukuda et al. 1997, DiMario, 2001).
As illustrated in this case report, the suspicion of an
incipient epileptic syndrome must always be excluded.
Although children with Dravet syndrome could have seizures provoked by water immersion, the most frequent,
initial clinical presentation of a long-lasting febrile convulsion, the evolution of the seizures and development is
completely different from that of HWE (Ceulemans et al.
2004).
Other types of epilepsy with onset at this age such as
benign myoclonic epilepsy of infancy or benign familial or
non-familial, infantile seizures (Watanabe-Vigerano syndrome) are not associated with seizures provoked by
water-immersion.
Febrile seizures are, as a rule, provoked by fever and are
mostly generalised tonic-clonic convulsions. Rare cases of
aquageneous urticaria might also be a kind of HWE
(Mouterde et al. 1997).
Treatment consists of avoiding contact with warm water. In
some cases, a good result may be obtained with conventional anti-epileptic drugs or oral prophylaxis with benzo-
Epileptic Disord Vol. 10, No. 1, March 2008
diazepines before taking a hot bath (Mouterde et al. 1997,
Ioos et al. 2000, Sharma et al. 2002, Incecik et al. 2004).
The prognosis for hot water epilepsy is usually favourable.
There is mostly spontaneous remission and psychomotor
development remains normal (Argumosa et al. 2002).
Rather exceptionally, there may be an evolution to nonreflex epilepsy (Satishchandra et al. 1988). M
Legend for video sequence
The video shows a seizure provoked by placing the
child in a warm bath.
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