Intractable
yawning caused by foramen magnum meningioma in a
patient with neurofibromatosis type
2
Bayri Y, Tanrikulu B, Bayrakli F, Koç
DY, Dagçinar A
Department of Neurosurgery,
Marmara University Faculty of Medicine,
Istanbul
Yawning is a stereotyped event seen in all
vertebrates. Neural networks in the brainstem,
autonomic nervous system, hypothalamus, and
limbic system may be involved in the physiology
of yawning. Tumor-related yawning has rarely
been reported. [1],[2] We
present a rare case of a patient with a
meningioma at foramen magnum who presented with
intractable yawning. After total removal of the
tumor, the recurrent yawning resolved
completely.
A 17-year-old girl was admitted with
complaints of headache for 2 years. Her
neurological examination revealed no abnormality
other than hearing deficit in the right
ear.
Her magnetic resonance imaging (MRI) of the
brain revealed hydrocephalus, bilateral
vestibular schwannomas with the right one being
23 * 25 * 22 mm and the left one,14 * 8 * 6 mm,
in size. She also had a falcine meningioma, that
was 13 * 10 * 11 mm in size. Due to the presence
of these bilateral vestibular schwannomas, she
fulfilled the diagnostic criteria of
neurofibromatosis type 2 (NF2).
The patient underwent a
ventriculo-peritoneal shunt and had radiosurgery
for the right vestibular schwannoma and the
falcine meningioma. The left vestibular
schwannoma was left untreated as it was small in
size and she had completely preserved hearing on
that side.
Fourteen months after the administration of
stereotactic radiosurgery, the patient was again
admitted to the hospital with the complaint of
an involuntarily tilt of her head to the right
side. Any attempts to correct the head position
precipitated intractable yawning. She denied any
sleeping disorder. The patient's Epworth
Sleepiness Scale was 2/24. Her cranial MRI,
revealed an additional meningioma, 10 * 12 * 15
mm in size located toward the right side of the
foramen magnum extending down to the C1 level
[Figure 1]. The patient had no
additional neurological deficits.
Figure 1: Preoperative
axial, coronal and sagittal T1 weighted contrast
MRI of the patient showing homogenously
enhancing mass lesion at the right side of
foramen magnum extending down to C1 level. The
lesion has shifted medulla oblongata
She was operated through a posterior
approach using a dorsal midline skin incision
between the external occipital protuberance and
spinous process of C2. The tumor was dissected
meticulously from the surrounding structures,
and total excision was performed. It was
histopathologically diagnosed as a psammomatous
grade 1 meningioma.
The patient had no additional neurological
deficits after surgery. A postoperative
cranio-vertebral junction MRI confirmed complete
tumor removal [Figure 2]. Following
surgery, the patient was able to hold her head
in the normal anatomical position. The
phenomenon of intractable yawning on correction
of head position had resolved completely. At
follow-up after 9 months, there was no
recurrence of the yawning phenomenon.
Yawning involves wide opening of the mouth
with concurrent deep inspiration, followed by a
slow expiration. This complex and
well-coordinated process resembles a classical
reflex because once it starts, it is completed
without any influence from an external stimuli.
[2] However, yawning is not a simple
reflex because it has a complex spatio-temporal
organization with contributions from facial and
respiratory muscles, and other component
systems.
It is proposed that the complex neuronal
network that regulates yawning may be located in
the brainstem reticular formation. This is
supported by the observation that yawning also
occurs in anencephalic newborns in whom only
medulla oblongata exists as a brain structure.
[3]
In our patient, the symptom was probably
associated with the meningioma that was located
at the foramen magnum and C1 level. The
involuntarily tilting of her head to the side of
the tumor relieved pressure on medulla. This
made her involuntarily maintain that position of
the head prior to tumor removal. Yawning started
with correction of the head position because
this maneuver, in all likelihood, stretched the
medulla and increased tumor pressure on it. In
addition, yawning disappeared immediately after
surgery and did not recur.
Figure 2: Postoperative
axial, coronal and sagittal T1 weighted contrast
MRI of the patient showing complete removal of
the lesion
There are two cases in the literature that
report the phenomenon of intractable yawning
associated with brain tumors. One is related to
a hemangioblastoma of the IVth ventricle, and
the other is related to a mature teratoma of
supramedial cerebellum. [1],[4]
Our unique case reports yawning triggered by an
extra-axial lesion at the level of foramen
magnum.
