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Ph i l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y Vo l . 31 no. 2 Ju l y – de c e m b e r 2016
CASE REPORTS
Phi l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y 31
ABSTRACT
Objective: To present an unusual cause of pulsatile tinnitus, presenting in a young adult suering
from chronic recurrent foul-smelling discharge from the same ear.
Methods:
Design: Case Report
Setting: Tertiary National University Hospital
Patient: One
Results: A 24-year-old woman presented with pulsatile tinnitus on a background of chronic
recurrent foul-smelling discharge. Clinico-radiologic ndings seemed consistent with a
glomus tympanicum coexisting with chronic suppurative otitis media with cholesteatoma.
She underwent tympanomastoidectomy with excision of the mass. Histopathologic evaluation
revealed the mass to be granulation tissue.
Conclusion: Pulsatile tinnitus is rarely associated with chronic middle ear infection. Granulation
tissue arising at the promontory may mimic glomus tumors when accompanied with this
symptom. Despite this revelation, it would still be prudent to prepare for a possible glomus tumor
intraoperatively so that profuse bleeding and complications may be avoided.
Keywords: tinnitus, pulsatile; otitis media, suppurative; glomus tympanicum
Tinnitus is a complaint experienced by around 10% of the general population. Pulsatile
tinnitus, on the other hand, is an uncommon complaint that occurs in 4% of patients experiencing
tinnitus.1 It is a symptom rarely associated with chronic middle ear infection. The aim of this report
is to present one such case of pulsatile tinnitus in a patient with chronic recurrent otitis media.
CASE REPORT
A 24-year-old woman presented at the outpatient clinic with a three-year history of
continuous unilateral pulsatile tinnitus of the left ear. She also experienced recurrent episodes of
otorrhagia associated with ear manipulation from the same ear, as well as recurring foul-smelling
ear discharge for six years, with gradually worsening ipsilateral hearing loss. There was also
occasional otalgia, headache, and vertigo.
Granulation Tissue Mimicking a Glomus Tumor
in a Patient with Chronic Middle Ear Infection
Kimberly Mae C. Ong, MD1
Patrick John P. Labra, MD1
Rosario R. Ricalde, MD1
Criston Van C. Manasan, MD2
Jose M. Carnate, Jr., MD2
1Department of Otorhinolaryngology
University of the Philippines -Philippine General Hospital
Manila, Philippines
2Department of Laboratories
University of the Philippines -Philippine General Hospital
Manila, Philippines
Correspondence: Dr. Rosario R. Ricalde
Department of Otorhinolaryngology
Ward 10, Philippine General Hospital
University of the Philippines Manila
Taft Avenue, Ermita, Manila 1000
Philippines
Phone: (632) 554 8400 local 2152
Email: chitricalde@yahoo.com
The authors declare that this represents original material, that
the manuscript has been read and approved by all the authors,
that the requirements for authorship have been met by each
author, and that each author believes that the manuscript
represents honest work.
Disclosures: The authors signed disclosures that there are no
nancial or other (including personal) relationships, intellectual
passion, political or religious beliefs, and institutional aliations
that might lead to a conict of interest
Presented at the Philippine Society of Otolaryngology Head
and Neck Surgery Interesting Case Contest. June 30, 2016.
Unilab Bayanihan Center, Pasig City.
Presented at the UP-PGH Department of ORL Interesting
Case Presentation. April 1, 2016. Philippine G eneral Hospital,
Manila.
Philipp J Otolaryngol Head Neck Surg 2016; 31 (2): 31-35 c Philippine Society of Otolaryngology – Head and Neck Surgery, Inc.
Creative Commons (CC BY-NC-ND 4.0)
Attribution - NonCommercial - NoDerivatives 4.0 International
Ph i l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y Vo l . 31 no. 2 Ju l y – de c e m b e r 2016
32 Phi l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y
CASE REPORTS
A
B
Figure 1. Otoscopic Findings: A. Normal otoscopy, right ear. B. 20% attic
perforation with gross cholesteatoma; remaining tympanic membrane appears
thickened, er ythematous with violaceous discoloration of the anteroinferior
quadrant; ear canal was lled with purulent discharge admixed with whitish
debris which was removed.
Figure 2. Plain CT scan of the temporal bone. A. Coronal cut of the right side
showing a shar p scutum with no soft tissue density in the epit ympanum. B.
Coronal cut of the left side showing blunted scutum, erosion of the ossicles,
soft tissue density within the epitympanum, and distinct sof t tissue mass at the
promontory (arrow).
Otoscopy revealed an attic perforation of the tympanic membrane
in the left ear with clinical evidence of cholesteatoma. There was also
note of a violaceous discoloration of the anteroinferior quadrant of the
tympanic membrane. (Figure 1)
Pure tone audiometry and speech testing revealed profound
hearing loss on the left ear with no recordable response at maximum
stimulus for both air and bone conduction. The right ear had normal
hearing acuity.
A non contrast-enhanced Computed Tomography (CT) scan of the
temporal bone revealed sclerosed mastoid air cells of the aected side
with well-developed air cells on the contralateral mastoid. (Figure 2) The
epitympanum of the aected ear was “lled with soft tissue density”
A
B
and showed “evidence of an osteolytic process with a blunted scutum
and eroded ossicles.” A soft tissue mass was noted in the promontory,
distinct from the soft tissue density in the epitympanum. In contrast,
the contralateral side showed well-pneumatized mastoid air cells
with a sharp scutum and absence of soft tissue densities within the
tympanic cavity. The internal carotid arteries on both sides were each
separated from their tympanic cavities by the carotid plates. Likewise,
both jugular bulbs were separated from the tympanic cavities by their
corresponding sigmoid plates.
A Magnetic Resonance Imaging (MRI) with contrast, requested to
assess the nature and extent of the mass, revealed a “well-dened,
enhancing T1-weighted and T2-weighted imaging-intermediate
Ph i l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y Vo l . 31 no. 2 Ju l y – de c e m b e r 2016
CASE REPORTS
Phi l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y 33
subcentimeter focus situated within the left tympanic cavity.” (Figure 3)
The said mass “did not exhibit restricted diusion on diused-weighted
imaging. The left mastoid air cells were sclerosed. Petrous pyramids
appeared normal and symmetrical. Contrast administration was not
followed by an abnormal rise of signal intensity within the nerve,
especially its intrameatal portion. There was loss of normal uid signal
in the left lateral semicircular canal” suggesting inltration.
She underwent canal wall down mastoidectomy, excision of the
middle ear mass, and meatoplasty. Intraoperatively, there was note of a
perforation in the attic retraction. A cholesteatoma was seen involving
the epitympanum extending into the aditus ad antrum and sinus
tympani. Bony destruction was seen at the epitympanum with the
ossicles missing; and at the sinus tympani where the stapes footplate
was replaced by a cholesteatomatous material over the oval window.
There was no evidence of cholesteatoma entering the oval window.
The tympanic segment of the facial nerve was dehiscent. The tegmen
tympani was intact and the sigmoid sinus was protected by a thick layer
of sclerosed bone. The mastoid tip was uninvolved as its air cells were
not developed. The middle ear mucosa appeared normal except for the
area surrounding the cholesteatoma.
A polypoid, weakly pulsatile mass was seen at the area of the
promontory, adjacent to Jacobson’s nerve. (Figure 4) The mass bled
slowly and continuously but decreased in size and in amount of
Figure 3. MRI with contrast of the internal auditory canal showing a well-dened mass (white
arrow) at the left tympanic cavity measuring 0.9 x 0.6 x 0.7cm (CC x W x AP). A. T1-weighted
image shows an isointense mass B. Gadolinium- enhanced T1-weighted image showing
heterogeneous enhancement C. T2-weighted imaging showing intermediate enhancement.
Figure 4. Intraoperative ndings: A. Removal of the cholesteatoma (asterisk) from the
epitympanum extending to the aditus ad antrum. B. Pulsating mass found at the cochlear
promontory (arrow).
Based on these clinico-radiologic ndings, our patient was assessed
to have a glomus tympanicum and chronic suppurative otitis media
with cholesteatoma of the left ear.
A review of systems revealed that she never had any episode of
palpitation, abnormal bowel movement, polyuria, polydipsia, weight
loss, or blood pressure spikes. To denitively rule out the possibility of a
catecholamine-secreting paraganglioma, a catecholamine screen was
performed which turned out normal.
A
B
C
Ant.
ASup.
Post.
Inf.
*
B
Ph i l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y Vo l . 31 no. 2 Ju l y – de c e m b e r 2016
34 Phi l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y
CASE REPORTS
bleeding with application of epinephrine. There was a stalk attaching
the mass to the promontory. The mass was excised with cupped forceps
and application of epinephrine-soaked cotton was sucient to control
bleeding.
Histopathologic examination showed granulation tissue formation
with inammatory cells composed primarily of plasma cells along with
lymphocytes and macrophages. (Figure 5) Immunohistochemistry for
kappa and lambda chains were both positive, indicating a polyclonal,
non-neoplastic plasma cell population. (Figure 6)
Resolution of pulsatile tinnitus was noted in the immediate post-
operative period. No symptom recurrences were noted until 3 months
after surgery after which, the patient was lost to follow up.
DISCUSSION
When pulsatile tinnitus accompanies the otoscopic nding of a
retrotympanic mass, three entities are considered—arterial anomalies
such as an aberrant internal carotid artery, an exposed jugular bulb, or
an intratympanic tumor, the most frequent being a glomus tumor.2-5
Radiological investigation can distinguish these three entities.
Glomus tumors, also known as paragangliomas, are blood-vessel-
rich tumors that arise from paraganglion cells of neuroectodermal
origin often located near nerves and vessels. Glomus tympanicum
is the term used to describe paragangliomas originating from the
middle ear, often found on the cochlear promontory along the inferior
tympanic nerve, or Jacobson’s nerve. The pulsations generated by
the tumor are transmitted to the otic capsule, resulting in increased
bone conduction and allowing the sound to be perceived more
intensely. Microscopically, a paraganglioma is typically composed of
well-formed cuboidal cell nests, or “Zellballen.” (Figure 7) Interspersed
between these nests are highly vascularized, brous septa. Each cell
has an abundant, granular and basophilic cytoplasm. None of these
features were seen in the lesion.
Figure 5. Photomicrographs of the intratympanic mass (Hematoxylin-Eosin, 400X) showing
granulation tissue (broblasts and new blood vessels) with an inammatory inltrate of plasma
cells and macrophages.
Figure 7. File photomicrograph of a paraganglioma (Hematoxylin-Eosin, 400X) showing typical
‘Zellballen’ formations. (UPCM-PGH Department of Pathology)
(Hematoxylin – Eosin, 400X)
Figure 6. Immunostaining for Kappa and Lambda chains (400X magnication) were both
positive.
(Hematoxylin – Eosin, 400X)
An estimated 3-4% of head and neck paragangliomas secrete
catecholamines.6 Catecholamine-secreting paragangliomas, when
undiagnosed, can result in serious perioperative morbidity and
mortality. It is therefore important to have a high index of suspicion to
reduce complications.
A review of literature associating pulsatile tinnitus with chronic
middle ear infection yielded limited results. Falcioni, Taibah, and Rohit in
20047 reported a case of pulsatile tinnitus as a consequence of residual
cholesteatoma causing sigmoid sinus compression in a patient who
underwent surgery nine years earlier. However, this was not evident
Ph i l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y Vo l . 31 no. 2 Ju l y – de c e m b e r 2016
CASE REPORTS
Phi l i P P i n e Jo u r n a l of ot o l a r y n g o l o g y -he a d an d ne c k Su r g e r y 35
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radiographically or intraoperatively in our patient. Several cases of
glomus tumors coexisting with chronic otitis media have been reported
both in the adult and pediatric population8-10 but to our knowledge,
pulsatile tinnitus caused by granulation tissue from chronic middle ear
infection has not been previously reported.
Granulation tissue is formed as part of the process that follows tissue
injury and inammation, such as is seen in chronic otitis media and
cholesteatoma. More frequently associated symptoms of granulation
tissue in the middle ear include recurrent foul-smelling ear discharge
and hearing loss. Tinnitus, when present, is usually non-pulsatile.
Histologically, it is characterized by the proliferation of broblasts,
angiogenesis or the formation of new thin-walled capillaries, loose
extracellular matrix and inammatory cells such as macrophages.11
According to Michaels, “it is usually particularly prominent in the
middle ear under the mucosa covering the promontory from which it
frequently protrudes into the external canal through a perforation of
the tympanic membrane, forming an aural polyp.”12 The angiogenesis
occurring within the tumor could have caused pulsations that
transmitted to the otic capsule resulting in pulsatile tinnitus similar to,
but with less intensity than a glomus tumor would have caused.
A glomus tumor would present with homogenous and intense
enhancement following administration of contrast material owing to its
high vascularity. Particularly, a glomus tympanicum should manifest as a
small discrete mass arising from the cochlear promontory, and conned
to the tympanic cavity.13 Granulation tissue on computed tomography
should present as a non-expansile soft tissue attenuation material.14
Some enhancement may also be expected, but not as intensely as a
glomus would. However, non-contrast enhanced imaging such as seen
in our patient would only show non-specic soft tissue density for either
of these two entities causing the confusion in diagnosis.
Cholesteatoma, like granulation tissue, should present a soft tissue
density on CT, but unlike granulation tissue, is associated with bony
erosion of surrounding structures and a blunted scutum similar to
what was seen in our patient.
As in the case of CT, a glomus tumor should also show intense
enhancement following contrast administration on MRI. Characteristic
of all paraganglioma would be multiple serpentine and punctate areas
of signal void variably distributed throughout the mass giving it the so-
called “salt and pepper appearance.”13,15 On the other hand, granulation
tissue should present with an intermediate signal on T1-weighted
imaging, and a hyperintense T2 signal with diuse enhancement
on delayed post-contrast enhancement.14 Table 1 summarizes the
dierences in MRI ndings between these two entities as well as
cholesteatoma. This suggests that glomus tumors should have similar
ndings as granulation tissue on MRI.
In retrospect, granulation tissue as a cause of pulsatile tinnitus in our
case would have been more parsimonious with the history of chronic
suppurative otitis media. Nevertheless, a high index of suspicion for
a paraganglioma should always be maintained, as the intraoperative
complications and profuse bleeding from the latter are much more
serious.
Table 1. Comparative MRI findings of Glomus, Inflammation and Cholesteatoma
T1-weighted MRI T1 – weighted MRI
with gadolinium
contrast
T2-weighted MRI
Glomus tumor14
Inammation/scar
tissue15
Cholesteatoma15
Hypointensity
Areas of signal
void (“salt
and pepper”
appearance)
Hypointensity
Hypointensity
Hyperintensity
Areas of signal
void (“salt
and pepper”
appearance)
Hyperintensity
Hypointensity
Peripheral rim
(matrix)
Hyperintensity
Areas of signal
void (“salt
and pepper”
appearance)
Hyperintensity
Hyperintensity
