Intractable
Yawning as a Predominant Symptom of Temporal
Lobe Ganglioglioma:
Case
Report and Review of Literature
Kutty RK, Alapatt JP, Govindan A
Abstract
Yawning, a physiologic reflex exhibited by
vertebrates, is seldom noticed as a symptom of a
disease. Not too often is a patient aware of it
as a symptom, unless it is of such a distressing
nature to seek attention. In this situation, to
distinguish between normal and abnormal behavior
would pose a diagnostic dilemma for the
attending physician. Intractable yawning has
been a presenting symptom of many pathologic
states such as stroke, epilepsy, and migraine.
Literature is sparse regarding intractable
yawning caused by tumors of the brain. Most of
the time, the etiology cited is the
infratentorial location of these tumors causing
compression of the brainstem and the centers
responsible for yawning. Intractable yawning as
a predominant symptom of supratentorial tumor is
rare. We present a case of an 18-year-old girl
who presented with abnormal yawning. On
evaluation, magnetic resonance imaging revealed
a tumor in the posterior part of the inferior
temporal gyrus. There was no significant
compression of the brainstem structures to
suggest this as a cause for her symptom. She
underwent a craniotomy and total excision of
lesion. Postoperatively, her symptoms improved
and her salvos of yawns ceased. The
histopathological examination revealed a
ganglioglioma of the temporal lobe. The present
case is unique as it is the only case reported
in the literature of a supratentorial tumor
causing abnormal yawning.
Résumé
Le bâillement, comportement
physiologique commun à tous les
vertébrés, est rarement
observé comme symptôme d'une
maladie. Il n'est pas trop fréquent qu'un
patient en prenne conscience comme un
symptôme, à moins que ce soit si
pénible que son attention ne s'y
fixe.
Dans cette situation, faire la distinction
entre un comportement normal et anormal pose une
difficulté diagnostique au médecin
traitant. Le bâillement
irrépressible a déjà
été noté comme
symptôme au cours de nombreux états
pathologiques tels que : accident vasculaire
cérébral, épilepsie et
migraine.
La littérature est pauvre en ce qui
concerne les bâillements
irrépressibles causés par une
tumeur du cerveau. La plupart du temps,
l'étiologie rapportée a une
localisation sous tentorielle entraînant
une compression du tronc cérébral
et des centres responsables du bâillement.
Le bâillement irrépressible en
tant que symptôme prédominant d'une
tumeur sus tentorielle est exceptionnelle. Les
auteurs présentent le cas d'une jeune
fille de 18 ans qui présentait un
bâillement anormal. Lors de
l'évaluation, l'imagerie par
résonance magnétique a
révélé une tumeur dans la
partie postérieure du gyrus temporal
inférieur. Il n'y avait pas de
compression significative des structures du
tronc cérébral pouvant
suggérer être la cause de son
symptôme. Elle a subi une craniotomie et
une excision totale de la lésion.
Après l'opération, ses
symptômes se sont améliorés
et ses salves de bâillements ont
cessé.
L'examen histopathologique a
révélé un gangliogliome du
lobe temporal. C'est le premier cas
rapporté dans la littérature d'une
tumeur sus tentorielle provoquant un
bâillement répété
anormal.
Introduction
Yawning is a physiologic reflex controlled
by a complex set of neurons situated in and
around the brainstem. There are many theories to
the origin of this natural phenomenon described
in the literature. Common to them, the
paraventricular nucleus describes special
mention as it is cited to be the integration
center of this multi-phased reflex. Often this
is accompanied by pandiculations, a phenomenon
whereby it is associated with stretching and
raising of arms. Rarely, this reflex is
unchecked in individuals in various diseased as
well as iatrogenic states. When this happens,
the patients throw salvo of yawns otherwise
known as "chasms." There are diverse definitions
of what constitutes abnormal yawning. Many
authors have described it ranging from 2/10 to
30/10 min. Regardless of the number, it leaves
one with no doubt as to what "abnormal" is, when
the patients seek attention for it. Among few
reports that cite a brain tumor as the etiology,
most all of them have been due to tumors in the
infratentorial compartment compressing the brain
stem. We encountered a supratentorial tumor in
the temporal lobe as the cause of intractable
yawning.
Case report
An 18-year-old girl presented with a history
of the absence of awareness which lasted for 1-2
min followed by bouts of abnormal yawning of 24
months duration. She used to yawn at least 20-30
times in succession after which she would have
mild headache followed by drowsiness. She had at
least 10-12 such episodes at irregular intervals
during a day. She was treated by a local
physician who initiated her treatment with
carbamazepine. She was not responding well to it
when clonazepam was added as the second drug.
There was a change in her semiology of symptoms
where by her symptoms of loss of awareness
subsided and she was left only with these bouts
of intractable yawning. She had a lot of weight
gain as an adverse effect of carbamazepine when
she was referred to us. She underwent magnetic
resonance imaging (MRI) which showed a
well-circumscribed lesion of about 2 cm _ 2 cm
in the posterior part of the right inferior
temporal gyrus [Figure
_[Figure1a1a-d]. The tumor was partly
cystic with an enhancing mural nodule. There was
no significant mass effect or any midline shift.
There were no areas of blooming on gradient
echo. There was no hydrocephalus. An interictal
electroencephalography (EEG) was taken which
revealed focal slowing over the right temporal
region. She had no previous history of seizures.
There was no history of sleep deprivation. On
examination, she was fully conscious and
oriented. Her Epworth sleep scale score was
2/10. She had no history of sleep apnea. She had
no cranial nerve deficit. There was no weakness
involving any of the limbs. She had only yawning
as the complaint which was witnessed during the
entire hospital stay. She underwent right
temporo-occipital craniotomy and total excision
of the tumor. The tumor was well-circumscribed
with well-defined arachnoid plains. It was
moderately vascular and was excised totally. A
histopathological examination showed her lesion
to be ganglioglioma [Figure _[Figure2a2a
and _andb].b]. Postoperatively, her
frequency of yawning decreased. We were able to
reduce the dosing of her anticonvulsants. She
was weaned off from each gradually by the end of
one year and by that time she was totally free
of her symptoms. MRI was repeated after 5 years
and there was no recurrence of the lesion
[Figure _[Figure3a3a-d]. She has
been on our follow-up for last 5 years, and she
has been symptom-free until now.
Discussion
awning as a sequelae to tumors of the brain
is rare. Almost all of them are reported to be
due to infratentorial tumors.[1,2]
Although the occurrence of this phenomenon with
supratentorial pathology such as stroke is
common[3,4] it has never been reported
with tumors in that region. A thorough search on
PubMed, with terms intractable yawning, brain
tumors, and supratentorial did not reveal any
results. Hence, we believe this is the first
case report of intractable yawning as the cause
of a supratentorial tumor.
The mechanisms underlying the occurrence of
yawning are arcane. There are at least three
neurogenic pathways that have been identified in
the induction and control of yawning. All of
them cite, the paraventricular nucleus of the
hypothalamus and their connections as the prime
integration unit of this circuit.[4]
Yawning has been described occurring as a
preictal, ictal as well as postictal phenomenon.
When occurring in the preictal phase, it has a
localizing value as it indicates its origin in
the nondominant temporal lobe.[5] Our
patient had a tumor involving the nondominant
temporal lobe. Jasper and Penfield were the
first to describe their patient with
diencephalic epilepsy in which their patient had
yawning and other dysautonomias in addition to
seizures. Our patient had only loss of awareness
other than intractable yawning. We believe that
this was itself an ictal seizure manifestation.
Unfortunately, due to the lack of video EEG or
intraoperative electrocorticography in our
institution, this was not done. Ictal yawning
has been reported before although
rare.[6,7] Most of the pathology
responsible for abnormal yawning reported so far
have been located in close proximity to the
brainstem.[1,2,8] In our patient, the
lesion was located in the posterior part of the
inferior temporal lobe. There was no mass effect
on the brainstem to attribute it to the cause of
yawning. The histology of the lesion in our
patient was ganglioglioma, a glioneuronal tumor
which is a leading cause of long-term epilepsy
associated tumor, also known as LEAT. The
intrinsic nature of cells of ganglioglioma
predisposes to epilepsy. Glioneuronal tumors are
composed of peculiar cellular components with
hyperexcitable neurons, functionally integrated
into excitatory circuitries, and neurochemical
characteristics that can be relevant for
epileptogenesis.[9]
Abnormal yawning also has been reported in
supratentorial nonorganic lesions such as
stroke. The etiology cited in such instances
have been that supratentorial lesions release
their inhibitory effect on the yawning centers
of brainstem.[9] The fact that her
symptoms were mitigated immediate
postoperatively denotes that it was infact, the
focus of seizure which manifested as intractable
yawning in our patient.
4. Walusinski
O. Yawning in diseases. Eur Neurol.
2009;62:180-7.
5. 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):311-4
7. 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.
9. Giulioni M, Marucci G, Martinoni M,
Marliani AF, Toni F, Bartiromo F, et al.
Epilepsy associated tumors: Review article.
World J Clin Cases. 2014;2:623-41.
