Meningioma related trigeminal neuralgia presenting as odontalgia -A case report

untitledMeningioma related trigeminal neuralgia presenting as odontalgia -A case report-
Department of Anesthesiology and Pain Medicine, Kyungpook National University Hospital, *Department of Dentistry, Kyungpook National University, Daegu, Korea
Kyung-Hwa Kwak, Jeong Eun Lee, Jae-kyung Han, Doo Youn Hwang, Min Ji Kim, Younghoon Jeon*, and Jin-Seok Yeo
Received: June 25, 2012.
Accepted: July 12, 2012.
and Pain Medicine, Kyungpook National University Hospital, 50, Samdeok
2-ga, Jung-gu, Daegu 700-721, Korea. Tel: 82-53-420-5876, Fax: 82-53-
420-5041, E-mail: [email protected]
Classical trigeminal neuralgia is characterized by recurrent attacks
of lancinating pain in the trigeminal nerve distribution, and no cause
of the symptoms can be identified, other than vascular compression.
This type of injury may rarely be caused by identifiable conditions,
including tumor in the cerebellopontine angle. If the patient is sus-
pected for secondary trigeminal neuralgia, further evaluation is
required to diagnose and treat correctly. We report a case of a
49-year-old woman with a 1-month history of facial pain, who was
initially misdiagnosed as odontalgia, and even treated with the
extraction of her molar teeth. This case with the review of secondary
trigeminal neuralgia may highlight the difficulties of diagnosis, and
the importance of early diagnostic imaging, when trigeminal
neuralgia occurs with a brain tumor. (Anesth Pain Med 2013; 8:
117-120)
neuralgia, Trigeminal neuralgia.
by recurrent attacks of lancinating pain in the trigeminal nerve
distribution. Though mechanism has not yet been demonstrated
accurately, it has been proposed that nerve is directly
compressed by either blood vessel or tumor and nerve demye-
lination caused by chemical or physical damages [1-4]. For
most patients with trigeminal neuralgia, the results of their
clinical examination, imaging studies and laboratory tests are
unremarkable. This type of trigeminal neuralgia is called
“classical trigeminal neuralgia” and no cause of the symptoms
can be identified other than vascular compression. Secondary
trigeminal neuralgia has the similar clinical symptoms, but
another underlying cause is responsible for the symptoms such
as tumors or multiple sclerosis [4]. If the patient is suspected
for secondary trigeminal neuralgia, further evaluation is requ-
ired to diagnose correctly.
odontalgia and complaining of persisting pain after teeth
extraction, eventually diagnosed for trigeminal neuralgia
which occurred secondarily by a cerebellopontine angle
meningioma.
A 49-year-old woman presented with left facial pain for 1
month. The pain began from mandibular molar teeth and was
extremely sharp as cut by knife. Her molar teeth were
extracted and antibiotic treatment was done under the
suspicion of dental caries and chronic osteomyelitis as causes
of her pain in a local dental clinic, but the pain was not
ameliorated. Then, as her first visit to our pain clinic, we
prescribed daily 600 mg of carbamazepine under the clinical
impression of trigeminal neuralgia. However, her pain still did
not subside.
The patient complained of continuous pain at the degree of
visual analogue score (VAS) 8 upon the maxillary and mandi-
bular branch of the trigeminal nerve and was unable to sleep
because of severe pain. The intensity of pain had increased
along non-harmful stimulation such as salivation. But, there
was no other neurologic deficit. We conducted mandibular
nerve block with 1% lidocaine and increased the dosage of
carbamazepine up to 900 mg for pain control, but the pain
118 Anesth Pain Med Vol. 8, No. 2, 2013
Fig. 1. A homogeneous enhanced mass was revealed in left cerebellopontine angle lesion on T1 enhanced brain MRI.
was not decreased. Since the pain was continuous and did not
respond to the treatment, further diagnostic evaluation inclu-
ding brain magnetic resonance imaging (MRI) was undergone
to rule out other causes for trigeminal neuralgia. Brain MRI
study revealed that a 2.5 cm homogeneous enhanced mass
was found in left cerebellopontine angle region on enhanced
T1-weighted image (Fig. 1). Then, the patient was transferred
to the department of neurosurgery and diagnostically confirmed
as meningioma. She was started on 100 mg of phenytoin
three times daily and the pain started to be reduced. The
patient discharged hospital after gamma knife radiosurgery.
After procedure, her pain was initially disappeared, but two
months later, she complained of pain of the same region at
the degree of VAS 4. Until now, recent MRI findings did not
show the evidence of meningioma recurrence, and her pain
was controlled with pregabalin 150 mg and phenytoin 100 mg
three times daily.
As trigeminal neuralgia is the most common facial pain
among people over 50 years old, 4−5 people among 100,000
people are newly diagnosed for a year and it is a bit more
common in women with the ratio of 1 : 1.5 for men and
women [1-3]. There has not been any particular genetic cause
about this disease; however, it is reported that 2-4% of those
who are diagnosed for trigeminal neuralgia tend to accompany
multiple sclerosis [1].
Although the pathophysiology of trigeminal neuralgia is not
yet demonstrated clearly, the compression from either dorsal
root entry zone or nerve root due to blood vessel or tumor
around trigeminal nerve is considered to be major cause. Local
pressure causes demyelination that leads to abnormal depolari-
zation resulting in ectopic impulses. This type of injury may
rarely be caused by aneurysm, arteriovenous malformation,
tumor, and arachnoid cyst in the cerebellopontine angle or by
a traumatic event. In effect, it was reported the patients having
brain tumor from brain MRI is 2−12% among those who are
diagnosed for typical trigeminal neuralgia [5,6]. Therefore, to
rule out ‘secondary’ trigeminal neuralgia, imaging study is
required for those who have symptoms inconsistent with
classical trigeminal neuralgia.
Van Kleef et al. [2] suggested 6 simple questions to diag-
nose classical trigeminal neuralgia and strongly recommended
brain imaging study to exclude secondary trigeminal neuralgia
if the pain is not consistent with these questions. Six questions
are presented here: 1) Does the pain occur in attacks? 2) Are
most of the attacks of short duration (seconds to minutes)? 3)
Do you sometimes have extremely short attacks? 4) Are the
attacks unilateral? 5) Do the attacks occur in the region of the
trigeminal nerve? 6) Are there unilateral autonomic symptoms?
As these questions suggested, trigeminal neuralgia is charac-
terized with a sudden, sharp and unilateral pain and it is
expressed with the sensation like lancinating, shooting, and
burning. It may involve one or more branches of the
trigeminal nerve, with the maxillary branch involved the most
often and the ophthalmic branch the least. Pain-free interval
exists between attacks. The attack can continuously occur when
the patient is awake, but it does not occur when sleeping.
Trigger areas are distinguishing factor and the attack is
provoked due to harmless stimulus like brushing teeth or
washing [3]. Since the patient in this case had persistent pain,
clinician should have suspected secondary trigeminal neuralgia
and should have carried out a brain imaging study.
On the other hand, the pain presented in this case should
be considered from a perspective of odontalgia, Although
odontalgia is dull in nature, it sometimes can be sharp and
tend to be increased with hot or cold stimulation or beating.
It is usually not continuous, better or worse as time passes
and can disturb sleep. Odontalgia mostly contains the iden-
tifiable causes like teeth decomposition, prosthetic appliance,
periodontal disease and teeth fracture. This can be removed
by local anesthesia of teeth [7]. More relevant to this case,
atypical odontalgia must be ruled out to diagnosis trigeminal
Kyung-Hwa Kwak, et alSecondary trigeminal neuralgia 119
neuralgia. Atypical odontalgia shows dull and continuous pain
aspect. Patient with atypical odontalgia have a history of
dental nerve treatment or teeth extraction. Trigger areas are
rare and it is usually occurred among middle-aged women
[7].
are meningioma, acoustic neuroma, pituitary adenoma, epider-
moid tumors, schwannoma and multiple sclerosis. Acoustic
neuroma is the tumor which has the highest frequency among
tumors located in the posterior cranial fossa and meningioma,
pituitary adenoma and schwannoma are commonly the tumors
located around central fossa; besides, epidermoid tumors rarely
can be the cause as well [8]. Benign tumors of the skull base,
most frequently meningiomas may result in compression of the
nerve root entry zone within the posterior fossa or the
trigeminal ganglion and peripheral nerves within the middle
fossa and cavernous sinus. Meningiomas are mostly benign
tumors that are more frequent in women than men and
develop with an increased incidence in older patients. Since
meningioma dose not usually invade into surrounding tissue
and appears clearly on brain MRI, it is recently treated with
stereotactic radiosurgery. It was reported that tumor recurrence
rate and complications are lower in the stereotactic radiosur-
gery group than the surgical group [9]. However, local tumor
control may not necessarily imply improvement in sympto-
matology, although stereotactic radiosurgery represents excellent
local control of benign brain tumors as a feasible noninvasive
option. Huang et al. [10] assessed the outcomes of 21 gamma
knife radiosurgery treatments for benign tumor-related trige-
minal neuralgia. Twelve patients attained pain relief without
medication, with 17 patients (81%) reporting improvement of
their pain following gamma knife radiosurgery. The mean time
to pain improvement was 3.7 months. However, 4 patients
subsequently experienced recurrent pain, yielding a rate of
durable relief (ranging from partial to complete alleviation) of
62% [10]. The patient presented in this case, also demonstrated
a relapse of pain after 2 month complete resolution with
gamma knife radiosurgery.
Carbamazepine is an iminostilbene derivative antiepileptic
drug [11] and the initial treatment of choice as drug therapy
for patient with trigeminal neuralgia. Dosages used have
ranged from 100 to 2,400 mg per day; however, most of
patients show improvement with 200 to 800 mg per day. If
carbamazepine is unsuccessful or provides only partial relief
options include adding a second agent or switching drugs.
Phenytoin, gabapentine baclofen, lamotrigine and topiramate be
added or substituted for carbamazepine [3]. Recent study on
pregabalin in trigeminal neuralgia represents pregabalin appears
to be effective in the trigeminal neuralgia. In an open label
study aimed to evaluate the efficacy of pregabalin in
trigeminal neuralgia, Obermann et al. [12] reported that 39 of
53 patients (74%) improved after 8 weeks with a mean dose
of 269.8 mg/day (range 150−600 mg/day) pregabalin: 13
(25%) experienced complete pain relief and 26 (49%) reported
pain reduction over 50%, whereas 14 (26%) did not improve.
Patient presented in this case responded to phenytoin partially
before gamma knife radiosurgery and recurrent pain after
procedure was controlled with phenytoin and pregabalin
combination.
Trigeminal neuralgia may be an initial presenting symptom
indicating a cerebellopontine angle tumor. Therefore, not only
dentist or clinician, but also pain specialist should have a good
knowledge of this disease and evaluation processes including
medical examination, radiological examination, and neurological
examination. If the patient has a symptom of atypical trige-
minal neuralgia, or there is a symptom inconsistent with 6
questions mentioned above, brain MRI should be considered
[2].
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