Received: 2 September 2019 Revised: 17 December 2019 Accepted: 20 March 2020

DOI: 10.1111/scd.12461

CA S E H I ST O RY R E P O RT

Orofacial pain secondary to acoustic neuroma—A case report

Juliana Araújo Oliveira1 Karina Matthes de Freitas Pontes2 Romulo Rocha Regis2
Tereza Nicolle Burgos Nunes1 Sérgio Araújo Holanda Pinto3 Lívia Maria Sales Pinto
Fiamengui2

1 FederalUniversity of Ceará, Fortaleza,

Ceará, Brazil
2 Department of Restorative Dentistry, Federal
University of Ceará, Fortaleza, Ceará, Brazil
3 Department of Clinical Odontology, Federal
University of Ceará, Fortaleza, Ceará, Brazil
Correspondence
Lívia Maria Sales Pinto Fiamengui, Depart-
ment of Restorative Dentistry, Federal Univer-
sity of Ceará, Fortaleza, Ceará, Brazil.
Email: livia_holanda_@hotmail.com
Abstract
Aim: The present study aimed to report a case of orofacial pain secondary to acoustic
neuroma (AN).
Methods and Results: A 66-year-old female presented with unilateral facial pain and
odontalgia. The pain was described as throbbing, dull, and constant. Tinnitus, hearing
loss, dizziness, and others symptoms were also present. Due to the characteristics of
the pain and clinical or radiographic findings, other possible diagnoses, such as tem-
poromadibular disorder, tooth-related pain, sinusitis, and primary headaches, were
excluded. Somatosensory tests for allodynia and hyperalgesia showed extraoral and
intraoral hypersensitivity. Magnetic resonance imaging revealed a lesion located on
the right cerebellopontine angle extending into and obliterating the internal auditory
canal and compressing the middle cerebral peduncle, the pons, and the cisternal seg-
ment of cranial nerve V. The patient was diagnosed with a brainstem tumor compatible
with AN and trigeminal neuralgia secondary to cranial nerve V compression.

Conclusion: Although uncommon, intracranial tumors should be considered during

orofacial pain evaluation to avoid iatrogenic treatment and delayed diagnosis.

KEYWORDS
acoustic neuroma, orofacial pain, trigeminal neuralgia

1 I N T RO D U C T I O N people per year.12 It is a benign brainstem tumor involv-

Orofacial pain is a recurrent cause of searching for dental
care;1 although most cases originate in the stomatognathic
system, there can be other causes involved such as headaches,
neuropathies, and intracranial tumors.1
Although uncommon, a large variety of facial symptoms
may be associated with intracranial tumors, and dentists
should be aware of these pathologies as possible underlying
causes for orofacial pain to avoid unnecessary treatment.2
Acoustic neuroma (AN), or vestibular schwannoma, is an
example of an intracranial tumor that may cause orofacial
pain.3–11 In the United States, its incidence is 1.2 per 100 000
ing abnormal growth and proliferation of Schwann cells that
most commonly involves the vestibular division of the eighth
cranial nerve supplying the inner ear.3–5 Although benign,
AN can lead to serious morbidity through compression of
vital structures such as cranial nerves V to XII and the
brainstem.4,5,13
The most typical symptoms associated with AN are hear-
ing loss and tinnitus, but disequilibrium, vertigo, dizzi-
ness, numbness or tingling in the face, headache and symp-
toms, such as temporomandibular disorders (TMD) may also
occur.2,3,12,14,15 Trigeminal neuralgia (TN) and trigeminal
neuropathy secondary to AN are also possible symptoms that

© 2020 Special Care Dentistry Association and Wiley Periodicals, Inc.

Spec Care Dentist. 2020;1–5. wileyonlinelibrary.com/journal/scd 1

2 OLIVEIRA ET AL.
occur due to direct tumor pressure on the trigeminal nerve or
due to a tumor pushing an artery in contact with the trigeminal
nerve.2,4,6-8,13,15
Idiopathic TN (ITN) is the most common form of cranial
neuralgia.16 It is a disorder unrelated to other pathologies
characterized by recurrent, unilateral, short-term shock-like
episodes of pain with abrupt onset and end confined to the
somatosensory distribution of one or more branches of the
trigeminal nerve.16–18 It is triggered by nonpainful stimuli
such as chewing, brushing teeth, washing the face, shaving,
speech, or touching the affected dermatome zone.18–20 Addi-
tionally, it may mimic an odontogenic pain.2,4,5 It is often mis-
diagnosed, and 33% to 65% of patients undergo unwarranted
dental interventions.21
Previous studies found that 14.3% to 27.2% of patients with
TN had associated intracranial structural lesions on magnetic
resonance imaging (MRI).22–24 Idiopathic and secondary TN
present with similar symptoms, and some authors have sug-
gested MRI for all patients diagnosed with TN to exclude
intracranial lesions.23,24
Few cases of TN secondary to AN mimicking, TMD or
odontogenic pain have been reported.3–5,8–11 Due to the possi-
ble symptoms, knowledge of this disorder is extremely impor-
tant to avoid delays in AN diagnosis and iatrogenic treatments.
Therefore, the aim of the present study is to report a case of
orofacial pain secondary to AN.
2 CA SE REPORT
A 66-year-old female was referred to our Orofacial Pain Ser-
vice with the chief complaint of daily facial pain on the right
side, more specifically the supraorbital and zygomatic area
extending to ala nasi. Intraorally, pain occurred in both the
superior and inferior dentoalveolar region and was accompa-
nied by odontalgia on the right maxillary second molar.
The facial pain was described as throbbing, dull, and con-
stant, with episodes of stabbing pain graded as 8 accord-
ing to a 0-10 Visual Analogue Scale (VAS). In the morning,
and while washing her face, talking, chewing and brushing
her teeth, it was described as intolerable, leading to avoid-
ing brushing her teeth. Other symptoms, such as ipsilat-
eral tearing and rhinorrhea, especially upon waking, tinni-
tus, pain in the temple area, gradual hearing loss, mild imbal-
ance during domestic activities, jaw locking or stiffness, were
also described. The patient also reported controlled diabetes,
hypertension, and anxiety.
The patient declared that the pain started when she was 59
years old. At that time, the pain was localized on the right
side of the tongue, mandible, and maxilla and was described
as mild, dull, and constant and more pronounced on the right
maxillary second molar. The patient reported several dental
evaluations, but all professionals declared no oral or dental
F I G U R E 1 Panoramic radiography
alterations were needed. At same time, the patient presented
to otolaryngologists complaining of hearing loss, but they also
declared no clinical treatments were needed. Over the years,
the pain and hearing loss gradually worsened.
During clinical examination, she did not present with tem-
poromandibular joint (TMJ) or muscle pain complaints dur-
ing manual palpation. Additionally, the right maxillary sec-
ond molar already had endodontic treatment and did not show
physical or radiographic alterations (Figure 1). Tests of allo-
dynia and hyperalgesia were performed using a cotton swab
and the tip of a dental explorer, respectively. Both stimuli were
used intraorally and extraorally. Hypersensitivity was detected
extraorally from below the right eye to the mandibular body
and intraorally in both superior and inferior dentoalveolar
regions.
Due to the medical history and clinical and radiographic
findings, other possible diagnoses, such as TMD, tooth-
related pain, sinusitis, and primary headaches, were excluded.
MRI was requested, in which an expansive T2-hypersignal
lesion measuring 3.3 × 3.3 × 2.5 cm was found located in the
right cerebellopontine angle, extending into and obliterating
the internal auditory canal and compressing the middle cere-
bral peduncle, the pons, and the cisternal segment of cranial
nerve V (Figure 2). The patient was diagnosed with a brain-
stem tumor compatible with AN and TN secondary to cra-
nial nerve V compression. Palliative treatment with 100 mg
carbamazepine twice a day was prescribed, and after 1 week,
the patient showed total pain remission. Next, the patient was
referred to a neurosurgeon.
3 DIS CUS S IO N
In the present case report, the patient presented with orofa-
cial pain secondary to AN with several symptoms that could
lead to misdiagnosis. The patient had complaints of otological
symptoms of hearing loss, tinnitus, and imbalance. According
to Toledo et al,25 otological symptoms are frequently asso-
ciated with TMD, with ear fullness being the most preva-
lent (74.8%), followed by otalgia (55.1%), tinnitus (52.1%),
OLIVEIRA ET AL.
F I G U R E 2 Contrast axial MRI showing an expansive lesion
located in the right cerebellopontine angle extending into the internal
auditory canal
vertigo (40.8%), and hearing loss (38.9%). Although frequent,
these symptoms should be analyzed with caution by a special-
ist. In addition to TMD, other possible diagnoses were consid-
ered, such as primary headache, more specifically paroxysmal
hemicrania, and sinusitis. Tooth-related pain, such is pulpitis
and periodontitis, was also ruled out.
Previous case reports related to orofacial pain secondary
to AN described pain as moderate to severe localized
in the maxilla and mandibular regions, with episodes of
shock-like pain compatible with TN.3,10,4,7,9 Other symptoms
reported were pain at preauricular and auricular3 regions,
tenderness or pain on the TMJ,3,9 masticatory muscles,3,10,9
toothache,3,10,4,5 facial numbness,7,5 burning symptoms,7,5,13
tinnitus,3,4,5 hearing impairment,3,10,5 and intermittent loss of
balance.3,10
AN has been diagnosed in patients of all ages, but the inci-
dence increases with age, peaking in the sixth decade of life
and is more common in women.12 An increased incidence of
AN in recent decades has been reported, but it has not been
determined whether these data reflect a real increase in tumor
incidence or just easier access to imaging diagnosis,26 such
as gadolinium enhanced T1-weighted MRI, the gold standard
for diagnosis and surveillance in patients with suspected or
known AN.27
It is hypothesized that neuronal involvement related to AN
occurs by mass compression of the nerve,7 typically com-
pressing the root near its entry into the pons, which induces
focal demyelination and is thought to trigger paroxysmal
ectopic discharge.28 Another possible mechanism suggests a
distortion of the contents of the posterior fossa with displace-
ment of the nerve root against a blood vessel or the skull
base.29,30
3
Common characteristics of neuropathic pain conditions
are changes in somatosensory sensitivity with loss of affer-
ent sensory function and the paradoxical presence of certain
hyperphenomena such as allodynia, hyperalgesia, wind-up-
like pain and after-sensations.31 Frequently, patients with TN
are able to identify pain triggers, such as laughing, chewing,
shaving, or brushing their teeth,32 a phenomenon known as
allodynia, when pain is evoked by innocuous stimuli.33
In the present clinical report, symptoms similar to TN
were present such as severe electric shock-like pain triggered
by innocuous stimuli. The patient also reported dental pain,
which could lead to an odontogenic pain misdiagnosis, but
no clinical or radiographic alterations were identified. Pain
of nonodontogenic origin mimicking dental pain has been
reported in cases of TN, AN, herpes zoster, diabetic odon-
talgia, and myocardial infarction.34 When facial pain worsens
with chewing or manipulation of teeth and/or gums, patients
often consult their dentist first.32
To provide information about mechanisms underlying
pain conditions, a standardized battery of Quantitative Sen-
sory Testing (QST) was developed to study somatosensory
function.35 Therefore, a simpler, faster, and more qualitative
clinical method to assess somatosensory testing has been pro-
posed, the Qualitative Somatosensory Testing (QualST). It
provides information, such as hyposensitivity or hypersensi-
tivity to touch, cold, and pinprick stimuli, covering the func-
tion of A𝛽-, A𝛿, and C-fibers.36 In the present study, QualST
using touch and pinprick stimuli were used. These tests were
performed on both painful and contralateral sides to observe
sensorial differences.
AN may be managed through three distinct treatment
options, observation, radiation, and surgical removal,37 and
the last one is the most common treatment modality.12 Con-
servative treatment, such as antiepileptics, may be indicated
for patients with few symptoms, small lesions, or who are not
able to undergo surgery.38
In this case report, after AN diagnosis using medical his-
tory, clinical examination and imaging exams, the patient
received palliative treatment with carbamazepine to alleviate
clinical symptoms and was referred to a neurosurgeon. Car-
bamazepine (200-1200 mg/day) is the treatment of choice for
TN,39 and its response is better in cases of ITN. Patients pre-
senting with secondary TN neuralgia are less responsive to
treatment.40 Due to carbamazepine’s poor tolerability, oxcar-
bazepine may be preferred.39
4 CONC LU SI ON S
AN is frequently associated with a wide range of symptoms,
which may lead to several diagnostic hypotheses. Facial
pain that worsens during function, headache, and otologic
symptoms are frequently present and found in individuals
4 OLIVEIRA ET AL.
with TMD. Dental pain commonly occurs secondary to caries,
periodontal disease, sinusitis and myofascial pain, but it may
also be a symptom of a variety of neuropathies. Thus, intracra-
nial tumors, such as AN, must be considered during orofacial
pain evaluation to avoid misdiagnosis and treatment delays.
The patient gave permission to use clinical information and
photographic material.
CONFLICT OF I NT E R E ST
The authors have no conflict of interest to disclose.
ORC ID
Lívia Maria Sales Pinto Fiamengui
https://orcid.org/0000-0002-2746-2219
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How to cite this article: Oliveira JA, de Freitas
Pontes KM, Regis RR, Nunes TNB, Pinto SAH,
Fiamengui LMSP. Orofacial pain secondary to acous-
tic neuroma—A case report. Spec Care Dentist. 2020;
1–5. https://doi.org/10.1111/scd.12461