Hot-Water Epilepsy in Children

Sharma, Ajay; Sankhyan, Naveen

doi:10.1007/s12098-021-03840-3

Cited by 4 publications

(4 citation statements)

References 6 publications

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“…Reflex epilepsies, such as hot water epilepsy and bathing epilepsy, are characterized by seizures triggered by specific stimuli, particularly involving water during bathing. Hot water epilepsy, a well-known variety that is frequent in South India, manifests a few key clinical characteristics: (1) triggering of seizures by pouring hot water, with temperatures ranging from 38°C to 55°C, over the head; (2) the absence of neurodevelopmental problems or detectable abnormalities in neuro-imaging for most affected children; (3) generally favourable outcome for the majority of children diagnosed with hot water epilepsy ( 15 – 19 ). But a less well-known infantile variety, primarily documented in France, exhibits several distinct clinical features: (1) Seizures commence before the age of 1 year, triggered by immersion in hot water at approximately 37.5°C; (2) Similar to the variant prevalent in South India, these children typically do not exhibit neurodevelopmental problems or abnormalities in neuro-imaging; (3) The prognosis for these children is also generally positive; (4) EEGs often reveal a focal onset of seizures, predominantly in the temporal region; (5) No other type of seizure was reported ( 20 ).…”

Section: Resultsmentioning

confidence: 99%

SYN1 variant causes X-linked neurodevelopmental disorders: a case report of variable clinical phenotypes in siblings

Ren,

Wu,

Zhou

et al. 2024

Front. Neurol.

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The SYN1 gene encodes synapsin I, variants within the SYN1 gene are linked to X-linked neurodevelopmental disorders with high clinical heterogeneity, with reflex epilepsies (REs) being a representative clinical manifestation. This report analyzes a Chinese pedigree affected by seizures associated with SYN1 variants and explores the genotype–phenotype correlation. The proband, a 9-year-old boy, experienced seizures triggered by bathing at the age of 3, followed by recurrent absence seizures, behavioral issues, and learning difficulties. His elder brother exhibited a distinct clinical phenotype, experiencing sudden seizures during sleep at the age of 16, accompanied by hippocampal sclerosis. Whole exome sequencing (WES) confirmed a pathogenic SYN1 variant, c.1647_1650dup (p. Ser551Argfs*134), inherited in an X-linked manner from their mother. Notably, this variant displayed diverse clinical phenotypes in the two brothers and one previously reported case in the literature. Retrospective examination of SYN1 variants revealed an association between truncating variants and the pathogenicity of REs, and non-truncating variants are more related to developmental delay/intellectual disability (DD/ID). In summary, this study contributes to understanding complex neurodevelopmental disorders associated with SYN1, highlighting the clinical heterogeneity of gene variants and emphasizing the necessity for comprehensive genetic analysis in elucidating the pathogenic mechanisms of such diseases.

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Section: Resultsmentioning

confidence: 99%

SYN1 variant causes X-linked neurodevelopmental disorders: a case report of variable clinical phenotypes in siblings

Ren,

Wu,

Zhou

et al. 2024

Front. Neurol.

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“…Some literature suggests that bathing epilepsy is different from hot-water epilepsy ( 1 ). Hot-water epilepsy is related to the temperature of the water (ranging from 38°C to 55°C) during bathing, and lowering the water temperature during bathing reduces seizure incidence or no longer induces epilepsy at all ( 11 ). Most reports of hot-water epilepsy are from India and Turkey where ritual bathing involves repeatedly pouring hot water over the child’s head, suggesting a genetic predisposition.…”

Section: Discussionmentioning

confidence: 99%

Video-electroencephalographic findings and clinical characteristics of bathing seizures in children

Kuang,

Liao,

Fang

et al. 2024

Front. Neurol.

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ObjectiveTo explore the electroencephalogram (EEG) and clinical characteristics of childhood bathing epilepsy.MethodsWe conducted a prospective summary of the clinical data from 10 children with bathing epilepsy who were admitted to Hunan Children’s Hospital from April 2019 to November 2023 and analyzed their EEGs and clinical characteristics.ResultsOur 10 patients included eight males and two females, with seizure-onset ages ranging from 4 months and 20 days to 14 months. Nine cases showed normal intellectual development, and one case manifested delayed development. The Video-EEG (VEEG) findings showed that nine cases exhibited normal background with no interictal epileptic discharge. The seizures were characterized by lip cyanosis, tachycardia or bradycardia, weakness, paleness, and loss of consciousness. Ictal EEG revealed rhythmic fast waves, spike waves, spike-slow waves, or slow and sharp-wave activity over the temporal region (eight cases) or the occipital and temporal regions (one case), finally evolving into a delta rhythm that lasted for 57–201 s. These children exhibited no seizures after discontinuing bathing and were not administered antiseizure medication. The interictal EEG of one case reflected mild slow background and focal interictal epileptic discharge; and her semiology was eyes gazing to right, with clonic movements of the right face and lips, lip cyanosis, bradycardia, and impaired consciousness. Ictal EEG showed spike–wave and spike-slow-wave rhythms over the left central, parietal, and temporal regions; these then spread to the left hemisphere, lasting for approximately 104 s. This patient did not exhibit bathing seizures after stopping her bathing but later experienced frequent spontaneous and drug-resistant seizures. The interictal EEG background slowed down, while focal epileptic discharge increased. Her intellectual development was significantly delayed, and a novel pathogenic mutation in the SMC1A gene, c.298+2T>C, was detected. She was diagnosed with developmental and epileptic encephalopathy.ConclusionA majority of children with bathing epilepsy in our study showed focal autonomic seizures accompanied by impaired consciousness. Stopping bathing could control the seizures and showed a good prognosis. A few infants manifested a poor prognosis, and we posit that bathing seizure rarely constitute the early manifestations of developmental and epileptic encephalopathy. VEEG findings and clinical features can also indicate the prognosis.

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“…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 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.…”

Section: Discussionmentioning

confidence: 99%

“…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‐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.…”

Section: Discussionmentioning

confidence: 99%

Hot water epilepsy: a video case report of a Caucasian toddler

Ceulemans

Koninckx²,

Garmyn³

et al. 2008

Epileptic Disorders

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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].

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Hot-Water Epilepsy in Children

Cited by 4 publications

References 6 publications

SYN1 variant causes X-linked neurodevelopmental disorders: a case report of variable clinical phenotypes in siblings

SYN1 variant causes X-linked neurodevelopmental disorders: a case report of variable clinical phenotypes in siblings

Video-electroencephalographic findings and clinical characteristics of bathing seizures in children

Hot water epilepsy: a video case report of a Caucasian toddler

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