Rare disease

CASE REPORT

Reflex gelastic–dacrystic seizures following
hypoxic–ischaemic encephalopathy
Rajesh Verma,1 Heramba Narayan Praharaj2
1

Department of Neurology,
King George Medical
University, Lucknow, Uttar
Pradesh, India
2
Department of Neurology,
CSM Medical University,
Lucknow, Uttar Pradesh, India
Correspondence to
Professor Rajesh Verma,
drrajeshverma32@yahoo.com

SUMMARY
Reflex or stimulus-sensitive epilepsies are uncommon
epileptic syndromes triggered by exogenous-specific
sensory stimulus or endogenous various mental activities.
Gelastic–dacrystic seizures are rare epileptic manifestations
characterised by ictal laughter and crying. Gelastic–
dacrystic seizures are commonly caused by hypothalamic
hamartoma but rarely described due to cortical dysplasia,
lesions of frontal and temporal lobes, tumours and vascular
malformations. We report a young woman who presented
with somatosensory-evoked gelastic–dacrystic seizures. This
patient had a positive history of perinatal insult
substantiated by MRI findings. Hypoxic–ischaemic
encephalopathy as the cause of gelastic–dacrystic seizures
has not been reported so far in the literature.

BACKGROUND
Reflex epilepsies are a group of epileptic syndromes
in which epileptic seizures are triggered by specific
sensory stimuli or certain actions of the patient.
Reflex seizures may clinically manifest as partial or
generalised seizures. Seizure may be provoked by
light, auditory, autonomic, somatosensory, olfactory
or other mental activities such as listening music or
reading.1
Gelastic seizures, characterised by bouts of laughter
constitute a rare group of epileptic disorders which is
commonly associated with hypothalamic hamartoma.2 Uncommon causes of gelastic seizures
reported in the literature are cortical dysplasia, lesions
of temporal and frontal lobes, astrocytomas of mammillary bodies and third ventricular papillomas.3
Dacrystic seizures characterised by ictal crying are also
rare epileptic events associated with hypothalamic
hamartoma, tumours, vascular malformations, mesial
temporal sclerosis or cerebral infarction.4
In this case report and videos we present a
patient exhibiting tap-induced gelastic–dacrystic seizures associated with hypoxic–ischaemic encephalopathy. Hypoxic–ischaemic encephalopathy as a
cause for reflexive gelastic seizures is not described
in the literature to the best of our knowledge.

Semiology of these seizures could be described as
brief tonic–clonic movements accompanied by
crying and subsequently bouts of laughter and
regaining consciousness within 1–2 min (video 1).
These seizures were not provoked by loud voice or
light. Perinatal history revealed delayed cry.
Milestones were mildly delayed with decreased
scholastic performance.
Examination findings disclosed mini-mental
status scoring of 27/30 with no focal neurological
deficits. Sensory system examination did not reveal
any abnormality.

INVESTIGATIONS
Investigations revealed random blood sugar 96 mg/
dL, serum creatine 0.4 mg/dL and haemoglobin 12 g
%. Cerebrospinal fluid measles antibody titre was
negative. Electroencephalography (EEG) showed
periodic lateralised epileptiform discharges over the
right hemisphere (figure 1). EEG video of the patient
demonstrated gelastic–dacrystic seizure with ictal discharges predominantly in the right parietotemporal
areas (video 2). MRI of the brain, T2-weighted and
fluid-attenuated inversion recovery (FLAIR) images
revealed altered signal intensity suggestive of gliosis
and volume loss involving bilateral temporoparietal
regions indicating hypoxic–ischaemic encephalopathy (figure 2). On the basis of these peculiar findings
somatosensory evoked gelastic–dacrystic seizures was
the diagnosis.

TREATMENT
Initial treatment was phenobarbitone (60 mg/day)
prescribed by the physician. She poorly responded
to therapy. She was then prescribed a combination
of sodium valproate (500 mg twice a day) and

CASE PRESENTATION

To cite: Verma R,
Praharaj HN. BMJ Case Rep
Published online: [please
include Day Month Year]
doi:10.1136/bcr-2013010506

A 15-year-old girl presented with a history of convulsions since 5 years. Initially she manifested with
abnormal intermittent jerky movements of the left
upper limb without loss of consciousness. Her
parents noticed falling of objects from the left hand
during these episodes. Interestingly these seizures
occurred whenever the patient had touched objects.
Frequency of these seizures increased with the time.
These seizures were triggered by touch or tapping.

Verma R, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010506

Video 1 Video of the patient exhibiting tap induced
gelastic dacrystic seizure.
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Rare disease

Figure 1 Electroencephalograph of the patient revealing periodic lateralised epileptiform discharges on the right hemisphere.
clonazepam (0.5 mg thrice a day) and once again with poor
results. Presently she is on lamotrigine (50 mg twice a day and
levetiracetam (500 mg twice a day) resulting in significant reduction of seizures.

OUTCOME AND FOLLOW-UP
At follow-up after 3 months there was more than 50% reduction
in seizures.

DISCUSSION
Despite development of the best monitoring technology, hypoxic–
ischaemic encephalopathy remains a serious condition that causes
significant mortality and long-term morbidity, particularly in
developing countries. Hypoxic–ischaemic encephalopathy is one
of the most important causes of neonatal seizures. These neonatal
seizures usually occur 12–24 h after birth. Approximately 10% of

Video 2 Video-EEG demonstrating ictal discharges in right temporal
area.
2

patients of hypoxic–ischaemic encephalopathy develop epilepsy at
the age of 3.5 years. Only some patients develop epilepsy after this
age.5 In a population-based epidemiological study in children
from Brazil, the authors found that symptomatic focal epilepsy
was the most common epileptic disorder and hypoxic–ischaemic
encephalopathy as its leading aetiological factor.6 Our patient presented with reflex seizures after a gap of 10 years of perinatal
insult. Reflex epilepsy is an epileptic disorder characterised by provoked seizures due to external stimulus and sometimes due to
internal mental mechanism. Various reflex stimuli mentioned in
the literature are visual, auditory, olfactory, somatosensory, vestibular, reading, music, internal thoughts and hot water baths.7 A case
was reported on somatosensory rub-evoked epilepsy originating
from the temporal lobe. In this study, the authors described a
patient developing seizures evoked by a touch or rub on the

Figure 2 MRI of the brain, T2-weighted fluid-attenuated inversion
recovery image showing bilateral gliotic lesions in the parietotemporal
areas suggestive of hypoxic–ischaemic encephalopathy.
Verma R, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010506

Rare disease
unilateral arm and shoulder. Single-photon emission CT (SPECT)
in the immediate postictal phase disclosed contralateral temporal
lobe origin.8 Our patient manifested with gelastic–dacrystic seizures. Gelastic epilepsy is an infrequent seizure disorder described
as stereotyped, unprovoked incongruous ictal laughter. The commonest cause of gelastic seizures is hypothalamic hamartoma,
although few reports mentioned other aetiologies, like frontal lobe
lesions, temporal lobe involvement, parietal lobe disorders and
focal cortical dysplasia of cingulate gyrus.3 A patient suffering
from gelastic seizures due to involvement of the right parietal lobe
has been reported. The seizures described were brief staring followed by smiling and laughing. Seizures originating from the right
parietal lobe were substantiated by ictal EEG and SPECT.9
Dacrystic seizures are characterised by ictal crying and rarely associated with gelastic component. Again it is commonly due to hypothalamic hamartoma but rarely dacrystic seizures can occur
because of tumours, vascular malformations, hippocampal sclerosis and cerebral infarction. In a recent publication a case with
dacrystic seizures due to left temporal lobe involvement was
reported. This patient had recurrent complex partial seizures with
crying that was reproduced during left carotid methohexital anaesthesia. The author concluded that localised networks involving
limbic structures may be involved in patients with dacrystic
seizures.10

Contributors RV made the hypothesis and HNP helped in drafting the final version
of the manuscript.
Competing interests None.
Patient consent Obtained.
Provenance and peer review Not commissioned; externally peer reviewed.

REFERENCES
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Learning points

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Cerullo A, Tinuper P, Provini F, et al. Autonomic and hormonal ictal changes in
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Neurophysiol 1998;107:317–22.
McConachie NS, King MD. Gelastic seizures in a child with focal cortical dysplasia
of the cingulate gyrus. Neuroradiology 1997;39:44–5.
Lopez-Laso E, Mateos Gonzalez ME, Camino LR. Giant hypothalamic hamartoma
and dacrystic seizures. Epileptic Disord 2007;9:90–3.
Robertson CMT, Finer NN. Educational readiness of survivors of neonatal
encephalopathy associated with birth asphyxia at term. J Dev Behav Pediatr
1988;9:298–306.
Sampaio LP, Caboclo LO, Kuramoto K, et al. Prevalence of epilepsy in children from
a Brazilian area of high deprivation. Pediatr Neurol 2010;42:111–17.
Lee SA, Choi EJ, Kang JK. Reflex epilepsy induced by playing oriental card or board
games. J Clin Neurol 2006;2:262–7.
Hsieh CY, Chiou NJ, Wu YG, et al. Somatosensory rub evoked reflex epilepsy of a
temporal lobe origin. Neurol Sci 2011;32:297–9.
Shin HY, Hong SB, Joo EY, et al. Gelastic seizures involving the right parietal lobe.
Epileptic Disord 2006;8:209–12.
Tatum WO, Loddenkemper T. Crying with left temporal lobe seizures and Wada
testing. Epilepsy Behav 2010;18:303–5.

▸ Gelastic–dacrystic seizures are rare epileptic manifestations
characterised by ictal laughter and crying.
▸ Gelastic–dacrystic seizures are commonly caused by
hypothalamic hamartoma.
▸ Hypoxic–ischaemic encephalopathy can rarely cause
gelastic–dacrystic seizures.

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Verma R, et al. BMJ Case Rep 2013. doi:10.1136/bcr-2013-010506

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