CHOLESTEATOMA IN CHILDREN: CLINICAL PARTICULARITIES AND THERAPEUTIC OUTCOMES OF 57 CASES

M. Benzamit, N. Djarmouni, I. Meguellati, R. Adjissa, B. Khababa, L. Allouche, D. Benmessahel, M. Bouache, Y. Aguida

Setif University Hospital, ENT Department, Setif, Algeria

Introduction

The term ‘cholesteatoma’ is attributed to Johannes Müller in 1838. Radicals ‘chole’ and ‘steat’ refer to the cholesterol crystals and fatty acids found within this tumor, the suffix ‘oma’ to any neo-formation tissue characterizing a tumoral appearance.14

Poorly named, cholesteatoma of the middle ear is defined by the abnormal presence in the cavities of the middle ear of a keratinizing squamous epithelium (skin) endowed with a potential for scaling, migration and erosion.

This epidermis, except in cases of congenital cholesteatoma, always originates from the deep part of external acoustic canal (EAC).58 According to Gray it is ‘wrong skin in the wrong place’.1,911

Within this debate, we have considered, for many years, that cholesteatoma in the child, compared to that in the adult, is special. This feature goes for everything from diagnosis to therapeutic management and it would be related to the specific characteristics of the child itself. Cholesteatoma in this age is characterized by:

An extremely difficult diagnosis. Hearing loss is usually unnoticed and otoscopic examination is often difficult due to the pusillanimity of the child.

The occurrence of a pneumatized mastoid which is a subject of much debate.1216

A high aggression, responsible for a larger extension in the cavities of the middle ear and a greater number of complications.

The difficulty of surgical treatment. In fact, even a drastic well-performed procedure will not restore the ear to normal once and for all. In addition, despite the surgeon’s experience, cholesteatoma in children has a high tendency to recur (recurrence and residual).1719

In this study of 57 cases of acquired cholesteatoma of the middle ear of the child followed up at our department, clinical and paraclinical data and therapeutic modalities have been carefully recorded and analyzed. We tried to highlight the elements of choice of a therapeutic strategy and to chart the pathway for a preventive approach covering both pre-cholesteatoma states and the cholesteatoma itself, once declared.

Materials and methods

This is a longitudinal-type study on 57 cases of acquired cholesteatoma of the middle ear of children recruited and followed at the ENT department of Ferhat Abbas University of Setif (Algeria) between January 2004 and December 2008.

The study population is represented by 53 children whose age cut-off point is 16 years at diagnosis. The aim of this work is the study of clinical and paraclinical particularities, therapeutic approach and the evolution of this disease in the pediatric population.

Were carefully recorded: age, gender, history, reasons for consultation, examination of both ears, CT scan, the audiometric assessment, the localization and extension of the cholesteatoma, assessment of the lesions, and the surgical procedures and results.

In short, we had to deal with 389 variables. These data were recorded, entered and analyzed using the IBM software SPSS 11.5 (SPSS: Statistical Package for the Social Sciences).

Results

The average age is 12 years ranging from four to 15 years with a peak incidence at age 13. In fact, 77% of children were under 11 when they underwent the first surgical treatment. There were 32 boys and 25 girls (sex ratio 1.2).

In addition, low socio-economic status, unemployed father, large families with many dependent children, a mostly rural residence far from the health care centers are all factors identified in our study and characterize much of our cases. Personal otological history was often reported including either acute otitis media (49.1%) or serous otitis media (22.8%).

The time from the onset of the first clinical signs to the date of the treatment (S1C1) is also very important, but very difficult to specify. The results are impressive, showing extremes ranging from 32 to 131 months, therefore an average of 75 months. This result confirms, once again, the insidious development of cholesteatoma or sometimes even the carelessness of the parents.

As for reasons for consultation, the first sign is largely dominated by the fetid otorrhea (96.5%) characteristic of the disease. Hearing loss is well behind (66.7%). Cophosis was not detected in any case, neither in the ear to be operated nor in the controlateral ear. Indeed, only one case had a bone-conduction hearing loss at 40 dB pre-operatively. The mean pre-operative air-bone gap is 27.1 dB.

Otorragie, an alarming sign motivating an urgent consultation, is noted in more than 38% of cases and is often the revelation of a polyp occupying the ear canal, characterized by the suffering of the lining of the tympanic cavity (Fig. 1).

At other times, cholesteatoma was revealed immediately by a complication. This situation applied to one out of four children (14 cases; 24.6%). The most common complication was acute mastoiditis (15.8%) with different modes of externalization, retro-auricular exteriorization was even the most frequent complication. Dizziness was noticed in 3.5% and facial palsy also in 3.5%, which regressed dramatically after medical treatment and surgery. We also noted a case of meningitis in a girl of four years, and a lateral sinus thrombosis and subdural abscess in the posterior fossa in a boy of 14.

After physical examination, no differences in the affected side were noted. There were more lesions of the pars tensa than of the pars flaccida, and these lesions were the cause of cholesteatoma in 65% of cases.

In more than 70% of cases the controlateral ear was diseased, with cholesteatoma in seven cases (a rate of 12.2%). We also noted the existence of an ear infection with effusion and/or tympanic retraction in over 36% of cases, a considerable figure that raises interest for a preventive action.

A CT scan remains the radiological examination of choice. A CT-scan was made pre-operatively in 84% of cases and in only 14% of cases during post-operative monitoring. It enabled us to study the anatomy of the middle ear cavities (a good pneumatization of the mastoid was detected in more than 61% of cases) and to make an assessment of the lesions of cholesteatoma (localization, extension and damage caused by it)(Fig. 2).

All patients were operated on first-hand. Overall, 81 operations were performed. Of these, 34 were performed using the closed technique (59.6%) and 23 using the open technique (40.4%). Nineteen cases, however, required a second or third time of revision surgery, and in five cases the closed technique of the first time has been changed into a canal-wall-down (CWD) technique.

Whenever possible, the closed technique was always attempted first. However, for particular situations, the open technique was used. These situations were either anatomical (mastoid eburnation and/or prolapses of the meninges or the lateral sinus: two cases), or clinical (complications: 11 cases), or even an important extension of cholesteatoma removal of which was considered difficult (nine cases). There was also the risk of patients not following up due to low socio-economic level or remoteness from health facilities (one case).

image

Fig. 1. Polyp of the external auditory canal of a left ear. A. Coronal cut of CT scan of the left ear, lysis of the descending branch of the uncus with prollabing aspect of tissue mass through the tympanic membrane into the lumen of the external acoustic meatus. Note the denudation of the tympanic portion of facial canal and that of the lateral semicircular canal. B. Oto-endoscopic aspect with rigid optic 0, showing the polyp at the bottom of the external acoustic meatus. C. Coronal cut of CT scan of the right ear, in the same child. Integrity of the ossicular chain, good ventilation of the middle ear cavities. Note the invagination of the pars tensa which is visible on clinical examination. D. Oto-endoscopic aspect with rigid optic 0, showing the poster superior retraction of the pars tensa which remains controllable and self-cleaning. E. Part of the cupular epitympanum. P: Polyp; T: Eardrum; Pe: Perforation of the posterior superior part of the pars tensa; MAE: External acoustic meatus; CSCL: lateral semicircular canal; CT: computed tomography; AIS: incudostapedical joint; R: Retraction; MS: myringosclerosis; MM: Handle of malleus.

The closed technique is a masto-antro-atticotomy transmastoid with transcanal atticotomy with a dual approach: through the duct, and transmastoid. The posterior tympanotomy was not done systematically, but it was performed in 67.6% of cases in response to the imperatives of excision of cholesteatoma. A second operation was performed in 15 cases among 34 operated by closed technique; this represents 44.1%, with an average period of 14.9 months. A third operation was necessary in two cases.

Ossiculoplasty was done whenever possible during the same operation, using the ossicles of the patient, usually the incus. In the absence of these materials or if they were embedded in cholesteatoma, the cortical mastoid bone was used instead. The cartilage (conchal or tragal) was not only used for reconstruction, but also doubled to ensure sound transmission in the open technique.

Over an average period of four years (three to five years) the overall recurrence rate (recurrence and residual) was approximately 45.6%, with more recurrence after the closed technique than after the open technique.

The mean post-operative air-bone gap was 23.3; this means an average gain of 3.9 dB compared to the average pre-operative air-bone gap of 27.1 dB. The difference is statistically significant at Khi square test (p < 0.001). A better functional outcome is obtained with ICW than with CWD and with an intact stapes than with a destroyed stapes.

image

Fig. 2. Some aspects of the different lesions observed CT. 1 and 2. Coronal and axial CT scan cut respectively, a right ear showing a cholesteatoma with extensive denudation of the lateral semicircular canal, the denudation of the meninges of the middle cerebral fossa. lysis of the scutum and the ossicles chain. Note also the denudation of the second portion of the facial canal. 3 and 4. CT of both ears of the same patient, we note the importance of pneumatization of both mastoids: right without cholesteatoma and left side with cholesteatoma, which was extended and digitiform at the operation. A: antrum; SL: Lateral sinus; CSCL: lateral semicircular canal; V: Vestibule; M: malleus; E: incus; C: tympanic cavity.

Discussion

G.J. Duverney (1648–1730) reported in 1683 local accumulation of steatoma in and around the skull base.20 Cruvelhier in 1829 named this a pearly tumor and it was Johannes Müller (1838) who named it cholesteatoma.24

Cholesteatoma was and is until today a subject of endless controversy mainly concerning its etiopathogenic mechanisms and modalities of surgical management.

In the literature, cholesteatoma in the child is often treated separately and considered special compared to that of adults. Our study enabled us to appreciate its clinical features, laboratory findings, evolution and the terms of its therapeutic management.

This is a disease of the second childhood occurring in a particular surroundings influenced by Eustachian-tube dysfunction and the upper respiratory tract infections very common at this age. Low socio-economic status and remoteness of health facilities were also reported.21,22

The average age is even higher than the upper age limit set is high.18,2326 The male predominance is marked and emphasized by all (Selmane, Yahi, Zribi).2628

All authors agree that the first clinical sign is the fetid otorrhea (96.5%) which is very characteristic and immediately attracts attention towards cholesteatoma especially in children.15,26,2830 Hearing loss is well behind (66.7%) and is often unnoticed for two reasons: firstly, this is often unilateral and secondly, the child cannot express this symptom.

Cholesteatoma revealed by a complication was noted in one child out of four. The most common complication was acute mastoiditis exteriorized in the post-auricular region. In the literature, this rate varies from 0 to 52% with relatively low rates in the so-called industrialized countries: 0 for Gillot and Tos,30 4.5% for Darrouzet et al.14 and higher rates elsewhere: 27% Prescott et al. in South Africa,25 33% for Rachidi in Mo-rocco28 and 46% for Cruz in Brazil.30

In Algeria, more specifically in its capital Algiers, Selmane26 and Yahi27noted rates of about 10.4% and 12.7%, respectively, while Benchaoui in Constantine (300 km east of Algiers) in her thesis on complications of cholesteatoma in all ages, noted rates between 47.9 and 52.1%.31 Thus, it seems quite clear that the development level of a country’s socio-economical conditions, remoteness from health facilities and level of education and awareness are very influential in the increase of the incidence of this disease, but also in when this disease makes its first appearance by a complication.

In terms of otoscopic data, the postero-superior quadrant of the pars tensa is the most common starting point of cholesteatoma found in both the study of our series (64.9%) and the literature (83% for Darrouzet,14 65.5% for Mishiro,17 43.6% for Schilder et al.32).

We encountered many difficulties in interpreting these data due to a significant inhomogeneity between different authors linked to the following points: 1) The lack of a clear boundary between retraction pocket in the pre-cholesteatoma state and cholesteatoma; 2) Each author related his own specific otoscopic aspects to a physiopathologic mechanism.

The status of the controlateral ear is very important to clarify. In fact, otitis media with effusion and tympanic retraction were noted respectively in 17.5 and 19.2% of cases in our study. Darrouzet noted 12 and 17%. For Lerosey, however, it was six and 25% of cases.14,16 Bilateral cholesteatoma was found in 12.8% of our cases. In the literature it varies from 0.8% for Selmane26 to 18.5% for Prescott.25 For Schilder it is bilateral in 15% of cases.30 This bilateral involvement further complicates the therapeutic management.

The treatment of cholesteatoma remains exclusively surgical because the main goal is total eradication of the disease. The second goal, once overlooked, has become as important as the first one: preservation and even restoration of hearing.

There is still no consensus on the question what to use: closed technique or open technique. The current trend in the literature is to respect the normal architecture of the middle ear, which implies a closed technique. Although some authors have performed the open technique, in their studies we always find arguments against the choice of this technique. For many others, the attitude is rather eclectic, depending on each separate case (Charachon, Darrouzet, Lerosey, Sanna et al., Tos, Smyth).12,14,16,18,30,33 Indeed, each technique has advantages and disadvantages. In our department, a closed technique was always attempted first. The current trend especially in children is a closed technique with mastoid obliteration.

In the vast majority of cases cholesteatoma is digitiform: 70% in our series, 60% for Yahi27 and 51% for Darrouzet.14 It is also very extensive, despite numerous classifications used in the literature (Sanna and Zini, Selmane, Saleh and Mills).18,26,30 In our study, we noticed that some anatomical regions were more involved than others, e.g., the epitympanum with three compartments (the anterior half the time, the lateral in more than two thirds of cases, but especially in the posterior in over 98% of cases); followed by the aditus which is almost always affected by cholesteatoma, while the antrum was affected in 87.7% of cases. The rest of the mastoid air cells was infected in 49.1% of cases.

It should be noted that cholesteatoma in children is known to affect especially the atrium,14,34,35 and is difficult to remove because it invades the tympanic cavity, particularly the retrotympanum. Indeed, the sinus tympani was invaded in 73.7% of our cases and the facial recess in 89.5% of cases.

In our series, in cases in which the ossicular chain was intact, the ossicle most affected was the incus, particularly its long process (72% lysis). In the literature an unbroken chain was observed in different percentages from one study to another. This rate varies from 9.7% (Silvola et al.) to 67% (Stangerup et al.).25,29,3641 Actually, it is higher in the European studies than in other studies, which suggests a rapid diagnosis and especially an early treatment of the disease before it destroys the ossicles.

Concerning function and reconstruction, the most interesting lesion to note is that of the superstructure of the stapes, 40.4% in our study. It was more than 76% for Prescott et al.25 in South Africa and only 15% for Stangerup et al.41 This will determine the ossicular reconstruction and predict the functional outcome.

The destruction of the ossicular chain associated with the lesion of the inner ear, especially the lateral semicircular canal (10.5% of cases) and that of the facial canal (19.3%) all show an aggressive cholesteatoma and also a long-lasting evolution of this disease (Fig. 3).

image

Fig. 3. Around the cholesteatoma. A. Epitympanic cholesteatoma with spontaneous atticotomy in a left ear, the stapes is present alone, there is also a lysis of its head (*). Enlargement of a spontaneous atticotomy (**) has led to better control of the extension of cholesteatoma, there is a denudation of the entire second portion of the facial nerve (NF). B. Erosion of the lateral semicircular canal without opening the membranous labyrinth (*): here the matrix of cholesteatoma is dissected. MAE: External acoustic meatus; LTM: Ttympano-meatal flap; NF, VII: Facial nerve; CSCL: Lateral semicirculair canal; FCM: Middle cranail fossa; Ad A: Aditus.

Ossiculoplasty was performed in 63 cases of 81 cases operated (any technique and time combined) of which 82.5% of cases at the first time. In almost all our cases, we had to use only autologous material because of the unavailability of ossicular prostheses. Actually, we prefer the ossicles of the patients (incus, malleus) if they can be used, otherwise the mastoid cortical bone or the cartilage can be shaped as needed.

Audiometric results: the mean post-operative air bone gap was 23.2 dB, indicating an overall improvement of 3.9 dB compared with the mean pre-operative air-bone gap of 27.1 dB. The difference is statistically significant at Khi square test (p < 0.001).

With the closed technique, the average post-operative air-bone gap is 19.3 dB, while it is in the range of 31.4 dB in the open technique. Similarly, with an intact stapes the mean post-operative air bone gap is 21.1 dB, while it is in the range of 27.3 dB when the stapes was destroyed (all techniques). We note, in light of these data and those of the literature, the hearing result is better with the closed technique than with an open technique and also with an intact incus as compared to a destructed one.14,16,23,26,32,34,37,4045

Anatomical result: whatever the procedure, open or closed, zero risk does not exist; a residual or recurrent cholesteatoma is found with various techniques, according to the study and the authors.

Residual cholesteatoma is newly formed from a matrix left in place voluntarily or involuntarily, while recidivism is a treatment failure which results in the formation of a new cholesteatoma, often on a retraction pocket, e.g., tubal dysfunction frequently reported in children.

Some authors19,34,3742 report about recurrence without identifying whether it is residual or recurrence. Silvola uses the term re-cholesteatoma,3739 others calculate the rate of recurrence and residual from the total number of operated ears, regardless the technique used. Finally there are those who report only the results of the revised ears. This caused a certain inhomogeneity in the comparison of results.

In our series, the overall recurrence (recurrence and residual) was 45.6% of cases (calculated by the total number of series); residual cholesteatoma in 26.3% and recurrent cholesteatoma in 21%.

In recent publications,1417,23,24,34 rates of residual cholesteatoma vary from 3.2 to 64%, while those of true recurrence vary from 8 to 30%. The risk of recurrence (residual recurrence) requires prolonged monitoring. If the emergence of a residual cholesteatoma is exceptional beyond five years,14,4042,46 recurrence may be more insidious and late,19,46,47 delayed recurrences after five years represent 30% of cases,42,46 whatever the technique performed. Yahi, in her study, found cases of recurrence at five and eight years after surgery,47 Austin up to 15 years.41

Schraff found that 90% of residual cholesteatomas occur during the first five years42 and Mishiro noted a recurrence rate even higher than the post-operative follow up is prolonged.17

While it is true that there is no consensus for the schedule of monitoring or its duration, the majority agrees that monitoring should be at least ten years,16,37 regardless the technical procedure used.

For others (Ayache, Schilder, Kaylie)11,32,48 this monitoring should be for life because the mystery of the etiopathogeny of this disease is still not resolved, also the monitoring of hearing is mandatory. Magnan noted that a new surgery after a gap of ten years is not seldom (6% of cases).46

To sum up, the risk of recurrence is always present, especially in children, due to the particularity of cho-lesteatoma in this age (digitiform, extensive, destructive) and the particularity of the child itself (particular immunity, adaptative disease, tubal dysfunction). This means life-long monitoring is mandatory, if not indispensable, at least annually. This monitoring is done at the clinic (otoscopic examination under a binocular microscope and oto-endoscope). Radiology is becoming indispensable, especially the new MRI techniques.

Conclusion

Cholesteatoma in the child is special. The large clinical latency and its misdiagnosis complicate not only the task of the surgeon, but also the prognosis of this threatening disease with a high potential of recurrence whatever the technique used.

The best anatomical and functional results are obtained with the closed technique, but this does not eliminate the high rates of recurrence. In fact, prevention is crucial. Efforts should be focused not only on the diagnosis and management of pre-cholesteatoma, but also on the cholesteatoma itself once diagnosed. This may reduce damage in the cavities of the middle ear. In other words: ‘Early diagnosis and treatment provide the best opportunity for eradication’.1

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Address for correspondence: Dr Benzamit Makhlouf, Service ORL et Chirurgie de la face et du cou, Clinque ORL – Ophtalmologie, CHU de Sétif 19000, Algerie. benzamit@yahoo.fr

Cholesteatoma and Ear Surgery – An Update, pp. 229–236

Edited by Haruo Takahashi

2013 © Kugler Publications, Amsterdam, The Netherlands