Article Type : Research Article
Authors : Boudhar H, Laraqui ON, lahjaouj M, loudghiri M, Bijou W, Oukessou Y, Abada RL, Rouadi S, Roubal M and Mahtar M
Keywords : Bilateral cholesteatoma; Chronic otitis media; Ossicular erosion; Canal-wall-up; Hearing preservation; Diagnostic delay; Surgical outcomes
Introduction:
Bilateral middle ear cholesteatoma is a rare but severe form of chronic otitis
media, characterized by keratinizing squamous epithelium in the middle ear,
leading to complications such as hearing loss, facial paralysis, and
intracranial involvement. Despite extensive research on unilateral cases,
bilateral cholesteatoma remains understudied, particularly in resource-limited
settings. This study aims to analyze its epidemiological, clinical, and
therapeutic particularities.
Materials
and Methods: A retrospective study was conducted into our department between
(2021–2025) involving 54 patients with surgically confirmed bilateral
cholesteatoma. Data included demographic characteristics, clinical
presentation, imaging (CT scans), surgical techniques (canal-wall-up vs.
canal-wall-down), and postoperative outcomes. Statistical analysis was
performed using SPSS.
Results:
The cohort showed a male predominance (55.5%) and a mean age of 17 years, with
33.3% aged 10–20. Diagnostic delay averaged 10 years, and complications
included meningitis (3.7%) and facial paralysis (1.85%). CT scans revealed
ossicular erosion (81.5%) and tegmen tympani erosion (44.4%). Closed techniques
were preferred (77.5%), yielding a 7.4% recurrence rate vs. 1.85% for open
techniques. Hearing improvement averaged 5–32 dB, with 27.3% achieving a Rinne
gap <20 dB.
Discussion:
Bilateral cholesteatoma poses unique challenges due to its aggressive nature
and need for bilateral hearing preservation. The high diagnostic delay
underscores gaps in early detection, while conservative surgical approaches
balanced recurrence risk with functional outcomes. CT imaging proved critical
for preoperative planning. Limitations included lost-to-follow-up (61.1%) and
short follow-up duration (22 months).
Conclusion:
Bilateral cholesteatoma requires tailored surgical strategies and rigorous
follow-up to mitigate recurrence. Future efforts should prioritize early
screening, advanced imaging (e.g., MRI), and standardized protocols in similar
settings.
Bilateral
middle ear cholesteatoma is a rare but severe form of chronic otitis media,
characterized by the presence of keratinizing squamous epithelium in the middle
ear cavities, leading to potentially serious complications such as hearing
loss, vestibular dysfunction, facial paralysis, and intracranial involvement
[1]. Although unilateral cholesteatoma is well-documented, bilateral
cholesteatoma remains less studied, with distinct epidemiological, clinical,
and therapeutic features that warrant special attention. Epidemiological data
indicate that bilateral cholesteatoma accounts for 8% to 17% of cholesteatoma
cases, with a male predominance and a higher incidence among children and young
adults [2,3]. Its pathogenesis is multifactorial, involving theories such as
epithelial migration, metaplasia, papillary proliferation, and retraction
pocket formation due to Eustachian tube dysfunction [4]. Diagnosis primarily
relies on clinical examination and imaging, particularly computed tomography
(CT), which helps assess lesion extent and guide therapeutic decisions [5].
Treatment is surgical, with the goals of complete cholesteatoma eradication,
prevention of recurrence, and preservation or improvement of hearing. Surgical
techniques include closed (canal-wall-up) and open (canal-wall-down)
approaches, each with its own advantages and disadvantages [6,7]. However,
managing bilateral cholesteatoma is particularly challenging due to the need to
preserve hearing in both ears and the high risk of recurrence, estimated between
7.4% and 9.25% in reported series [3]. Our study aims to analyze the
epidemiological, clinical, and therapeutic particularities of this condition
while highlighting the challenges associated with its management in a setting
where long-term patient follow-up is often limited. The findings underscore the
importance of early diagnosis, tailored surgical strategies, and rigorous
surveillance to improve functional outcomes and reduce complications.
This
retrospective study was conducted in the ENT, Head and Neck Surgery Department
of the 20 august Hospital of Casablanca over a 5-year period (2021–2025). We
included 54 patients who underwent surgery for confirmed bilateral
cholesteatoma, representing 10.3% of the 524 cases of cholesteatomatous chronic
otitis media recorded during this period. Inclusion criteria consisted of a
diagnosis confirmed by clinical examination and computed tomography (CT), while
incomplete records or unilateral cholesteatomas were excluded. Data were
systematically collected from medical records using a standardized form,
including epidemiological parameters (age, sex, medical history), clinical
signs (otorrhea, hearing loss, complications), and results of complementary
tests. Otoscopic evaluation under a microscope allowed for the characterization
of tympanic lesions (retraction pockets, marginal perforations), while
pure-tone audiometry determined the type and degree of hearing loss. Temporal
bone CT scans, performed for all patients, provided essential information on
lesion extension, ossicular erosion, and bone destruction.
Regarding
treatment, two main surgical approaches were used: the closed technique
(canal-wall-up, CWU) with anatomical preservation and cartilage reconstruction,
and the open technique (canal-wall-down, CWD) via mastoidectomy. The choice of
technique depended on preoperative lesion extent, with reported recurrence
rates of 9–70% for CWU and 4–15% for CWD [6,7]. Ossiculoplasty was performed in
25% of patients during the first surgical intervention, primarily using
cartilage grafts or titanium prosthesis. Postoperative follow-up combined
clinical evaluations (assessing otorrhea or recurrence), audiometric tests
(measuring hearing improvement), and radiological examinations (follow-up CT
scans). Statistical analysis was performed using SPSS software, with appropriate
descriptive and comparative tests. This rigorous methodology allowed us to
comprehensively evaluate the specific characteristics of bilateral
cholesteatoma in our clinical setting.
The
analysis of 54 cases of bilateral cholesteatoma revealed a significant gender
distribution, with 30 males (55.5%) compared to 24 females (44.4%), yielding a
sex ratio of 1.25. Age distribution showed a clear predominance in pediatric
and young adult populations: 18 patients (33.3%) were between 10 and 20 years
old, with an overall mean age of 17 years (range: 10–66 years). The pediatric
population (<15 years) accounted for 14.8% of cases. Otologic history was
particularly frequent: 20 patients (37%) reported recurrent otitis media, and
19 (35.2%) had previously undergone tympanoplasty. A history of retraction
pocket reinforcement was noted in 8 patients (14.8%). The study revealed a
characteristic clinical presentation marked by chronic otologic symptoms.
Patients sought consultation after an average disease duration of 10 years
(range: 4 months to 30 years), with nearly 40% having a disease course
exceeding 10 years. The primary reason for consultation was fetid otorrhea,
present in 96.3% of cases, often bilateral (55.5%). Hearing loss, the second
most frequent symptom (72.2%), was also frequently bilateral (37%). Clinical
examination revealed objective signs: retraction pockets in 77.8% of cases
(predominantly attic retractions at 62%), marginal perforations (11.1%), and
external auditory canal polyps (20.4%). Severe complications, though rare,
included peripheral facial paralysis (1.85%) and meningitis (3.7%).
Systematic
complementary evaluation combined audiometry and imaging. Pure-tone audiometry
(performed in 50 patients) demonstrated conductive hearing loss in 70.4% of
cases (bilateral in 55.5%), mixed hearing loss (22.2%), and, less frequently,
sensorineural hearing loss (3.7%). Temporal bone CT scans, performed in all
patients, provided crucial information on lesion extent: ossicular erosion
(81.5% of cases), erosion of the scutum (75.9%), attic filling (46.3%), and
more severe findings such as tegmen tympani erosion (44.4%) or facial canal
involvement (3.7%). These precise radiological findings guided therapeutic
strategy (Figure 1) (Table 1,2). The surgical approach was tailored to each
case based on lesion extent. Initial unilateral surgery was preferred (74.1% of
cases), with a clear predominance of closed techniques (77.5%). For the 14
patients who underwent bilateral surgery (25.9%), the average interval between
interventions was 24 months. Frequent intraoperative findings included
inflammatory mucosa (78.6%), primarily attic involvement (71.4%), often
requiring antroatticotomy (55%). Ossiculoplasty, mainly type II, was performed
in 42.9% of bilateral cases, using cartilage grafts or titanium prosthesis.
Postoperative follow-up, available for 21 patients (38.9%), with complete
evaluations for 11 of them, showed after a mean follow-up period of 22 months:
satisfactory anatomical outcomes with an intact neotympanum in 54.5% of cases,
but recurrences in 36.4% of cases, more frequent after closed techniques. Functionally,
an average hearing improvement of 5–32 dB was observed, with 27.3% of patients
achieving a Rinne gap below 20 dB. These findings highlight the importance of
prolonged follow-up for early detection of recurrences.
CHL:
Conductive hearing loss; MHL: Mixed hearing loss; SHL: Sensorineural hearing
loss.
Figure
1: Audiogram Results.
The
results of our study provide significant insights into the epidemiological,
clinical, and therapeutic particularities of bilateral cholesteatoma. Our
series of 54 cases confirms and refines several essential aspects of this
complex ENT pathology. From an epidemiological perspective, the observed male
predominance (55.5%) aligns with data from the literature [2,3]. This
characteristic may be explained by several pathophysiological mechanisms. On
one hand, anatomical differences in the Eustachian tube typically narrower and
more horizontal in males could contribute to tubal dysfunction [4]. On the
other hand, hormonal factors might influence epithelial proliferation and
keratinization processes. The mean age of 17 years, with a peak incidence in
the 10–20 age group (33.3% of cases), confirms the early onset of this
pathology, likely linked to local immune system immaturity and the high
frequency of ENT infections in this age group. The clinical presentation is
particularly revealing. The mean consultation delay of 10 years significantly
longer than European data [8-12] highlights the challenges in accessing
specialized care in our case. This diagnostic delay likely explains the high
frequency of complications observed (5.55%), including potentially severe cases
such as meningitis (3.7%). The predominance of attic retraction pockets (62%)
supports the pathogenic theory of tympanic invagination as the primary
mechanism in bilateral forms [4,13]. These clinical findings underscore the
need to strengthen early screening efforts, particularly in children with
recurrent otitis media (Table 3).
The
analysis of paraclinical data provides major diagnostic insights. Audiometry
revealed more severe (70.4%) and more frequently bilateral (55.5%) conductive
hearing losses compared to unilateral forms, reflecting the often-symmetrical
involvement of the ossicular chain. CT scans, with a sensitivity of 85–93% for
detecting bone lesions [5], proved indispensable for assessing lesion extent.
Findings such as ossicular erosion (81.5%), scutum erosion (75.9%), and tegmen
tympani erosion (44.4%) directly influenced our therapeutic choices. From a
therapeutic standpoint, our conservative approach (77.5% closed techniques)
differs from some authors' recommendations [14-16] but appears justified by
several arguments. First, it allows better preservation of residual hearing
particularly crucial in bilateral cases. Second, it reduces immediate surgical
morbidity. However, this strategy has limitations, including a higher
recurrence rate (7.4% vs. 1.85% for open techniques) and requires rigorous
follow-up, often difficult to maintain in our context. Functional outcomes,
though modest (mean hearing improvement of 5–32 dB), remain encouraging. They
confirm that despite the aggressiveness of bilateral forms, satisfactory
auditory rehabilitation is achievable. The proportion of patients achieving a
Rinne gap <20 dB (27.3%) demonstrates the efficacy of modern ossiculoplasty
techniques [10]. Several methodological limitations must be acknowledged. The
high rate of lost-to-follow-up cases (61.1%) and the limited mean follow-up period
(22 months) restrict the generalizability of our long-term conclusions. The
absence of a unilateral control group prevents certain statistically
significant comparisons. Future improvements are multifaceted. The development
of postoperative MRI could enhance recurrence detection [5,17]. Less invasive
endoscopic techniques warrant evaluation for this indication. Finally,
optimizing follow-up protocols emerges as a priority to reduce the
lost-to-follow-up rate.