Excessive yawning has been reported in the
peri-ictal period preceding or following
seizures. We describe an exceptional case of an
elderly man with impairment of consciousness and
paroxysmal excessive yawning. We hypothesise
that this can be regarded as an autonomic
seizure originating from diencephalic/brainstem
structures, manifesting with yawning as an ictal
phenomenon.
We observed a case of an elderly man with
yawning as the main ictal seizure
manifestation.
Case presentation
An 86-year-old man presented with a history
of decreased responsiveness and recurrent falls
on a background of chronic general decline over
9 months. On hospital admission, investigations,
including routine blood tests, 24 h ECG and
blood pressure monitoring, showed no significant
abnormalities. A moderate degree of cerebral
cortical atrophy, but no space-occupying or
vascular lesion, was noticeable on a CT scan.
While on the ward, hospital staff observed the
patient having episodes of appearing 'absent'. A
routine EEG (Xltek EEG System, Oakville,
Ontario, Canada) was revealing and captured a
clinical event. About 12 min after the recording
started, the patient rested his head back on a
pillow and appeared still; 8 min later, the
patient became unresponsive. During the period
of unresponsiveness, which lasted 14 min,
clusters of yawns or single yawning episodes
were the most striking manifestation. During
this period, the EEG showed diffuse
electrodecremental activity (figure 1); a lead
II ECG showed a reduction in heart rate (mean 73
beats/min, compared to mean 93 beats/min before
and after the event). There was no clinical or
EEG response to external (visual, auditory,
tactile and painful) stimulation. Upon cessation
of this event, as the patient became alert and
responsive, the EEG returned to the original
resting state.
The patient was started on sodium valproate,
with a good clinical response: episodes of
appearing absent were no longer reported during
the period near to hospital discharge.
Unfortunately, in the longer term follow-up
period, the patient's general physical condition
declined markedly, with superadded intervening
clinical depression so that the patient's
relatives found not possible to report any of
those episodes.
Investigations
The key investigation was a routine EEG, as
it revealed changes occurring during a clinical
event. In fact, while the patient was
unresponsive, the EEG showed diffuse
electrodecremental activity; this abnormal
activity was different compared to the
background activity before and after the
clinical event. Hence, the EEG, showing abnormal
activity accompanying the clinical event, was
instrumental in guiding the diagnosis.
Differential diagnosis
Excessive yawning can occur in association
with clinical conditions such as brain tumours,
brainstem stroke, acute anterior circulation
stroke; however, our patient brain scan showed
no space-occupying or vascular lesion. Our
patient had no other neurological diseases such
as multiple sclerosis, amyotrophic lateral
sclerosis or migraine, conditions that have been
described in association with excessive yawning.
Our patient had a history of recurring episodes
of decreased responsiveness/appearing absent; we
captured one of these events, during which
excessive yawning occurred; for the duration of
this event, there were accompanying EEG changes.
All these features together are strongly
suggestive of a seizure disorder. Because of the
patient's change in level of alertness
(indicating brainstem involvement), the
paroxysmal yawning (with a putative yawning
centre in the brainstem) and the EEG
electrodecremental pattern (considered an EEG
seizure pattern of brainstem origin14), we
suggest that a seizure originating from the
brainstem structure is the most likely mechanism
underlying our patient's event.
Discussion
This is an exceptional case of prolonged
seizure with impairment of consciousness and
paroxysmal yawning. We hypothesise that this can
be regarded as an autonomic seizure originating
from diencephalic/brainstem structures,
manifesting with yawning as an ictal phenomenon.
Seizures accompanied by excessive yawning in the
periictal period have been described. Penfield
and Jasper were the first to report peri-ictal
yawning in two patients with autonomic
diencephalic seizures (but they did not mention
EEG). Peri-ictal yawning has been observed in a
child with infantile spasm, with the EEG showing
generalised decremental activity. Also,
peri-ictal yawning has been described in
patients with temporal lobe epilepsy, with
yawning preceding complex partial seizures (CPS)
and EEG showing interictal epileptiform
discharges,12 or with yawning during the
post-ictal period following CPS with or without
secondary generalisation and EEG showing
interictal epileptiform discharges. To the best
of our knowledge, there is only one reference
that describes a significant association between
ictal yawning and 3 Hz spike and wave
discharges. Golgie and Green studied a
7-year-old girl with petit mal; the frequency of
her seizures was 1 in 15 min, although it could
drop to 2/day. The girl was observed during a
prolonged EEG recording. They found that in 40 h
of total recording there were 351 yawns: 80
(23%) occurred during attacks and 271 (77%)
occurred during the resting periods; of the 80
yawns which occurred during attacks, 27 (34%)
occurred during spike and wave discharges and 53
(66%) occurred coincident with the onset or 20 s
preceding the onset of spike and wave
discharges.
We reported the case of an elderly patient
with yawning as the main ictal phenomenon during
a prolonged period of unresponsiveness. In 37
min of total EEG recording, 10 yawns were
observed, all of them occurring during the 14
min of unresponsiveness. Most likely, this could
be an autonomic complex partial seizure arising
from diencephalic/brainstem structures with
paroxysmal yawning. Cattaneo et al reported two
cases of brain stem stroke, with lesions
confirmed by MRI presenting with transient
excessive pathological yawning; the authors
hypothesised that the pathogenesis of excessive
yawning could be due to denervation
hypersensitivity of a putative brainstem yawning
centre. In our case, no identifiable brainstem
lesion was visible at brain imaging. However,
the involvement of brainstem structures is
unquestionable, considering the patient's change
in the level of alertness (unresponsiveness) and
the paroxysmal yawning. Nevertheless,
neocortical brain areas have a regulatory effect
on diencephalic and brainstem yawning centres,
as excessive yawning can occur in supratentorial
stroke. A neocortical contribution to the
excessive yawning in our case cannot be ruled
out. Moreover, the ictal EEG changes
(electrodecremental activity) observed during
the event in our patient are different than
those (spike and wave discharges) reported by
Golgie and Green. Diffuse electrodecremental
activity is considered an EEG seizure pattern of
possible brainstem origin, further corroborating
our hypothesis that our report could be an
exceptional case of a seizure originating from
brainstem structures. Our report is an addition
to the previous ones on excessive yawning and
emphasises the importance of this clinical
pathological manifestation in the elderly.
an other example of ictal yawning
References
1. Barbizet
J. Yawning. J Neurol Neurosurg Psychiatry
1958:21:203&endash;9.
2. Askenasy
JJ, Askensy N. Inhibition of muscle
sympathetic nerve activity during yawning. Clin
Auton Res 1996;6:237&endash;9.
3. Walusinski
O. Yawning: unsuspected avenue for a better
understanding of arousal and interoception. Med
Hypotheses 2006;67:6&endash;14.
4. Walusinski
O. Yawning in diseases. Eur Neurol
2009;62:180&endash;7.
6. Cattaneo
L, Cucurachi L, Chierici E, et al.
Pathological yawning as a presenting symptom of
brain stem ischaemia in two patients. J Neurol
Neurosurg Psychiatry 2006;77:98&endash;100.
7. Mogilnicka
E, Klimek V. Drugs affecting dopamine
neurons and yawning behaviour. Pharmacol Biochem
Behav 1977;7:303&endash;5.
13. Yankovsky
A, Andermann F,Dubeau F. Post-ictal forceful
yawning: an autonomic symptom in a patient with
nondominant hemisphere epilepsy. video-eeg
presentation Epilepsia 2005;46:s8; 23-34
14. Gastaut H, Roger J, Ouahchi S, et al. An
electroclinical study of generalize epileptic
seizures of tonic expression. Epilepsia
1963;4:15&endash;44.
15. Goldie
L, Green JM. Yawning and epilepsy. J
Psychosom Res 1961;5:263&endash;8.
Andy
OJ, M Jurko Diencephalic Seizure Appl
Neurophysiol 1983; 46; 62-67
Penfield
W, Jasper H Diencephalic autonomic seizures
Eplilepsy and the functionnal anatomy of the
human brain Little Brown et copany - Boston
1954
Penfield
W The influence of
the diencephalon and hypophysis upon general
autonomic function
Walusinski
O Yawning as a paroxysmal signof diencephalic seizures an original
observation
Specchio N,
Carotenuto A et al. Ictal yawning in a
patient with drug-resistant focal epilepsy:
Video/EEG documentation and review of literature
reports. Epilepsy Behav. 2011;22(3):602-605
Kuba R,
Musilová K, Brázdil M, Rektor
I. Peri-ictal yawning lateralizes the
seizure onset zone to the nondominant hemisphere
in patients with temporal lobe epilepsy Epilepsy
Behav 2010;19(3):311-314
