OTOSCOPIC CLINIC

Iatrogenic external auditory canal

cholesteatoma with mastoid erosion
Yen-Hui Lee, MD; Chih-Yu Hu, MD; Wen-Yu Chuang, MD; Kai-Chieh Chan, MD

Figure 1. Otoscopy displays a large, bulging mass in the posterosu-

perior aspect of the external auditory canal.

A 70-year-old man who had undergone postauric-
ular underlay myringoplasty to treat chronic otitis
media 10 years earlier presented with a 6-month
history of hearing impairment, aural fullness, and
occasional otorrhea of the right ear. Otoscopy revealed
a large, bulging mass on the posterosuperior aspect
of the external auditory canal (EAC); the tympanic
membrane was invisible (figure 1). Computed tomog-
raphy of the temporal bone revealed a right-sided, 2
× 2-cm soft-tissue mass in the EAC, with erosion of
mastoid air cells but a normal eardrum and middle
ear cavity.
Considering the postsurgical history and the site
of the mass, we diagnosed an iatrogenic EAC cho-
lesteatoma. A whitish, spherical mass was identified
on surgical exploration and was completely removed
through the prior postauricular incision (figure 2).
We subsequently performed canalplasty with EAC
defect reconstruction using fragments of conchal
cartilage. The pathologist’s report revealed histo-
pathologic features compatible with cholesteatoma.
No evidence of recurrent disease was present during
the 5-year follow-up.
Iatrogenic EAC cholesteatoma is a rare complication
developing after myringoplasty. The precise incidence
of the disease remains unknown. We speculate that
inversion or malpositioning of a tympanomeatal flap,
or unintentional implantation of epithelium during the
prior surgery, causes the subsequent defect.1,2
Small postsurgical EAC inclusion cysts or cholestea-
tomas are common and can be treated in the office via
“unroofing” of the cyst and good ear hygiene practices.3
However, large, complicated iatrogenic EAC choles-
teatomas usually require surgical management. The
technique chosen is generally based on the extent of
the disease and the surgeon’s preference, and it ranges
Continued on page 344

From the School of Medicine, National Taiwan University, Taiwan (Dr. Lee); the School of Medicine, Chang Gung University, Taiwan (Dr. Hu,
Dr. Chuang, and Dr. Chan); the Division of Otology, Department of Otolaryngology–Head and Neck Surgery (Dr. Hu and Dr. Chan); and
the Department of Anatomic Pathology (Dr. Chuang), Chang Gung Memorial Hospital, Linkou, Taiwan.

340 www.entjournal.com ENT-Ear, Nose & Throat Journal October-November 2018

 IMAGING CLINIC
Figure 2. Axial CT shows the dissected internal carotid artery
(arrow) and calcified stylohyoid ligament (asterisk).
might have been a harbinger of his impending cere-
brovascular accident.
Eagle Syndrome is an uncommon but well-described
entity with a nonspecific clinical presentation; more
benign manifestations include globus sensation,
dysphagia, facial neuralgias, throat pain, and cranial
neuropathies, for which a differential is extensive.1,2 In
the presence of an elongated styloid bone or stylohyoid,
diagnostic consideration is often given to Eagle syn-
drome, but it may have a more insidious presentation.
Elongation of the styloid also has been reported as a
cause for symptomatic carotid disease, including TIA,
Horner syndrome, eye pain, and cluster headache.3
Dissection associated with elongated styloids has been
reported in the neurology literature,4 but to the best of
our knowledge, no prior reports have demonstrated
TIA with a normal carotid and subsequent carotid
dissection during two separate clinical presentations.
Surgical and nonsurgical treatments for Eagle
syndrome have been reported. Medical treatments,
including carbamazepine, local and systemic steroids,
and NSAIDS have been used.4 Surgical options include
transpharyngeal resection, as originally favored by
Eagle, versus an extraoral resection.
References
1. Eagle WW. Elongated styloid process: Report of two cases. Arch
Otolaryngol 1937;25(5):584-7.
2. Werhun EL, Weidenhaft MC, Palacios E, Neitzschman H. Stylohyoid
syndrome, also known as Eagle syndrome: An uncommon cause of
facial pain. Ear Nose Throat J 2014;93(9):384-5.
3. Infante-Cossio P, Garcia-Perla A, Gonzáles-Garcia A, et al.
Compression of the internal carotid artery due to elongated
styloid process [in Spanish]. Rev Neurol 2004;39(4):339-43.
4. Zuber M, Meder JF, Mas JL. Carotid artery dissection due to
elongated styloid process. Neurology 1999;53(8):1886-7.
344 www.entjournal.com
OTOSCOPIC CLINIC
Continued from page 340
Figure 2. A spherical cholesteatoma sac is discovered during sur-
gical exploration.
from canalplasty with skin grafting for lesions confined
to EAC, to canalplasty or canal-wall-up mastoidectomy
with reconstruction of canal defects for lesions involv-
ing mastoid cells, to canal-wall-down mastoidectomy
for lesions with large wall defects exhibiting mastoid
erosion.4 If an EAC defect is to be reconstructed, as in
our case, nonretractable materials such as cartilage
make ideal grafts.
Myringoplasty is a commonly used and simple type
of middle ear surgery associated with a low risk of
postoperative, iatrogenic EAC cholesteatoma. How-
ever, it is important to ensure high accuracy when
repositioning the skin flap. In addition, delicate and
meticulous management of every step is essential to
avoid trapping or implantation of epithelial debris under
the skin flap or graft.
Our case emphasizes the need for long-term follow-up
because the average disease latency after otologic surgery
is usually more than one decade.1,5
Approval of this case study was obtained from the
Institutional Review Board of Chang Gung Memorial
Hospital.
References
1. Sweeney AD, Hunter JB, Haynes DS, et al. Iatrogenic cholesteatoma
arising from the vascular strip. Laryngoscope 2017;127(3):698-701.
2. Persaud R, Hajioff D, Trinidade A, et al. Evidence-based
review of aetiopathogenic theories of congenital and acquired
cholesteatoma. J Laryngol Otol 2007;121(11):1013-19.
3. Holt JJ. Ear canal cholesteatoma. Laryngoscope 1992;102(6):608-13.
4. Viswanatha B. External auditory canal cholesteatoma: A rare
complication of tympanoplasty. Ear Nose Throat J 2009;88(11):
1206-9.
5. Cronin SJ, El-Kashlan HK, Telian SA. Iatrogenic cholesteatoma
arising at the bony-cartilaginous junction of the external
auditory canal: A late sequela of intact canal wall mastoidectomy.
Otol Neurotol 2014;35(8):e215-21.
ENT-Ear, Nose & Throat Journal October-November 2018