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Case Report
Ceruminous Adenoma of the External Auditory Canal:
A Case Report with Imaging and Pathologic Findings
George Psillas,1Argyrios Krommydas,1Georgia Karayannopoulou,2
Kyriakos Chatzopoulos,2Jean Kanitakis,3and Konstantinos Markou1
11st Academic ENT Department, Aristotle University of essaloniki, AHEPA Hospital, 1 Stilponos Kyriakidi Street,
54636essaloniki,Greece
2Department of Pathology, Aristotle University of essaloniki, School of Medicine, 541 24 essaloniki, Greece
3Department of Dermatology, Ed. Herriot Hospital Group, Lyon, France
Correspondence should be addressed to George Psillas; psill@otenet.gr
Received September ; Revised October ; Accepted October
Academic Editor: Christian Koch
Copyright © George Psillas et al. is is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
Ceruminous adenomas are benign tumors that are rare in humans and present with a nonspecic symptomatology. e treatment
of choice is surgical excision. We present an -year-old woman who presented with a reddish, tender, round, so mass of the
outer third of the inferior wall of the le external auditory canal, discharging a yellowish uid upon pressure. Coincidentally,
due to her poor general condition, this patient also showed symptoms consistent with chronic otitis media, parotitis, and cervical
lymphadenopathy, such as otorrhea, through a ruptured tympanic membrane and swelling of the parotid gland and cervical lymph
nodes. e external auditory canal lesion was surgically excised under general anesthesia, utilizing a transmeatal approach. e
pathological diagnosis was ceruminous gland adenoma. e tumor was made of tubular and cystic structures and embedded in
a brous, focally hyalinized stroma. Immunohistochemistry conrmed the presence of two distinct cell populations. e luminal
cells expressed keratin , while peripheral (basal) cells expressed keratins /, S protein, and p. e apocrine gland-related
antigen GCDFP- was focally expressed by tumor cells. e postoperative course was uneventful and at the -year follow-up no
recurrence of the ceruminous adenoma was noted.
1. Introduction
Ceruminous adenoma (CA) is a benign neoplasm of cerumi-
nous glands, developing exclusively in the external auditory
canal (EAC) []. Contrary to felines and canidae [–],
it is rare in humans and poses a diagnostic problem for
the clinician, due to the variety of clinical presentations.
e wavering nomenclature existing for these lesions causes
additional confusion to pathologists and attending doctors
[]. We present herein a new case of CA and briey review
the relevant literature in order to delineate the salient clinico-
pathological features of this rare tumor [].
2. Case Report
An -year-old woman was referred to our department for
otalgia, yellowish discharge, itching, and hearing loss of the
le ear recurring over the last three months. She reported
intermittent fever, but neither tinnitus nor vertigo. She denied
previous trauma or surgery in this region and was not using
a hearing aid. Her past history included atrial brillation,
diabetes mellitus, arterial hypertension, and glaucoma. Oral
antibiotics and anti-inammatory medications, prescribed
elsewhere, were not eective.
Physical examination revealed a reddish, round, tender
somassoftheouterthirdoftheinferiorwallofthele
EAC, discharging a yellowish uid upon pressure; however,
a concomitant ruptured tympanic membrane and otorrhea
through this perforation were also found. No cranial nerve
palsy was detected. Swelling of the le parotid region and
bilateral cervical lymphadenopathy were also present. CT
scan of the temporal bone showed a cystic lesion on the
inferior cartilaginous part of the EAC but no temporal bone
lysis; the middle ear appeared to contain uid and swelling
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Case Reports in Medicine
Volume 2015, Article ID 359627, 3 pages
http://dx.doi.org/10.1155/2015/359627
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∗
F : CT imaging depicting otitis media of the le ear with
external ear canal obstruction (∗) due to the cystic lesion.
F : H/E ×. e tumor is made of tubular and cystic
structures.
of the parotid gland and cervical lymph nodes bilaterally
were also seen (Figure ). e audiogram revealed conductive
hearinglossoftheleear.epatientwastreatedwith
intravenous cefuroxime. A specimen of the discharge was
sent for bacteriological evaluation and turned out to be
negative for bacteria.
e patient underwent excision of one cervical lymph
node and ne needle aspiration (FNA) of the le parotid.
Histopathological examination of the lymph node ruled
out malignancy, being consistent with a granulomatous
process; however blood tests and thorough immunological
and rheumatological examination failed to conrm this
hypothesis. e FNA examination was nondiagnostic as it
showed normal salivary glands.
e EAC lesion was surgically excised under general
anesthesia, utilizing a transmeatal approach. Macroscopi-
cally the resection specimen was a fusiform skin sample
measuring 1.9 × 1.5 × 1 cm. Upon sectioning a . cm
cystic lesion was found in the subcutaneous tissue. Micro-
scopic examination showed an epithelial cystic lesion whose
wall contained two cell populations. e inner ones con-
sisted of middle-sized cylindrical or cuboidal cells with
an eosinophilic cytoplasm and round nuclei, arranged in
tubules (Figure ). e cylindrical cells occasionally showed
cytoplasmic projections in the lumens of the tubules. e
second cell population consisted of ovoid or spindle cells,
withelongatednuclei,whichwerearrangedinfascicles,small
solid groups, and rare tubules. Neither atypia nor mitotic
activity were observed. e tumor was embedded in a brous,
focally hyalinized stroma; it was partly surrounded by a
thin brous capsule and was not connected to the overlying
epidermis. Immunohistochemistry conrmed the presence
F : H/E ×. Two cystic-tubular formations lined by colum-
nar cells with an eosinophilic cytoplasm are seen.
F : Immunostain for keratin (×): luminal cells are
labelled.
of two distinct cell populations. e luminal cells expressed
keratin , while peripheral (basal) cells expressed keratins
/, S protein, and p. Several cells, mainly luminal,
showed membranous expression of CD/c-kit. p and Ki-
were expressed in about % and % of nuclei, respectively.
e apocrine gland-related antigen GCDFP- was focally
expressed by tumor cells. Based on these histopathological
and immunohistochemical ndings the diagnosis of cerumi-
nous gland adenoma was established (Figures –).
e postoperative course was uneventful and the excision
site was well healed. Moreover, the symptoms of otitis media
and cervical lymphadenopathy improved considerably. A
week later, the ear was free of secretions and the otalgia
disappeared. Aer days, the rupture of the tympanic
membrane had healed and hearing returned to its previous
condition. It is possible that, due to her age and poor
general condition, this patient presented with multifocal
infections, such as chronic otitis media, parotitis, and cervical
lymphadenopathy, which were rather coincidental to the
presence of CA.
No recurrence of the CA has been noted aer a -year
follow-up.
3. Discussion
AccordingtoMillsetal.[],CAarebenigntumorsthatare
rare in humans. ey usually aect patients over years old
Case Reports in Medicine
F : Immunostain for p protein (×): nuclear positivity is
seen in basal cells.
[, , –], but cases aecting adolescents exist []. However,
the view has been expressed that all ceruminous gland tumors
should be considered potentially malignant, as their clinical
behavior is not yet completely elucidated [, ].
Ceruminous adenomas appear as reddish polypoid
masses with either a smooth or ulcerated surface, which can
mimic a boil [, , , , ]. Although CA produce very
few symptoms, clinical manifestations such as hearing loss,
mild-to-moderate otalgia, and otorrhea have been reported
in some patients []. In our patient, the CA presented as
a smooth round mass discharging a yellowish uid upon
pressure; the other symptoms such as otalgia, otorrhea, fever,
and hearing loss were rather attributed to the concomitant
chronic otitis media.
Ceruminous adenoma shows immunohistochemical fea-
tures similar to those of normal cerumen glands, supporting
their origin from these glands [, ]. e dierential diagnosis
includes other tumors that develop in the EAC, namely,
exostosis, osteoma, eosinophilic granuloma, cholesteatoma,
cartilaginous choristoma, extra-adrenal paraganglioma, con-
genital cysts of branchial arch origin, ceruminous adenocar-
cinoma, pleomorphic adenoma, neuroendocrine adenoma
of the middle ear, and meningioma, and relies on care-
ful immunohistological examination []. e recommended
treatment is surgical excision of the lesion within free mar-
gins, which can ensure tumor-free survival in the long term
[,,,,].ereisnoevidenceofrecurrenceforthese
tumors in the literature, with a mean follow-up time of
years [, , , , ]. Our patient underwent total surgical
excision of the mass and showed no signs of recurrence up
to years later.
Consent
e patient provided written informed consent for publica-
tion of images and other information contained in this case
report.
Conflict of Interests
e authors declare that there is no conict of interests
regarding the publication of this paper.
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