Clinical analysis of secondary acquired cholesteatoma

    Clinical analysis of secondary acquired cholesteatoma Kazuhisa Yamamoto MD, Yuichiro Yaguchi MD, Hiromi Kojima MD PII: DOI: Reference:

S0196-0709(14)00131-8 doi: 10.1016/j.amjoto.2014.05.009 YAJOT 1398

To appear in:

American Journal of Otolaryngology–Head and Neck Medicine and Surgery

Received date: Revised date: Accepted date:

10 December 2013 19 May 2014 27 May 2014

Please cite this article as: Yamamoto Kazuhisa, Yaguchi Yuichiro, Kojima Hiromi, Clinical analysis of secondary acquired cholesteatoma, American Journal of Otolaryngology– Head and Neck Medicine and Surgery (2014), doi: 10.1016/j.amjoto.2014.05.009

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Clinical analysis of secondary acquired cholesteatoma

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Kazuhisa Yamamoto, MD, Yuichiro Yaguchi, MD, Hiromi Kojima, MD

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From the Department of Otorhinolaryngology, Jikei University School of Medicine, Tokyo, Japan

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A short running title: Secondary Acquired Cholesteatoma

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Financial disclosure: We have no financial disclosure and support.

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Conflict of Interest: None

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Send correspondence and reprint requests to: Kazuhisa Yamamoto, MD Department of Otorhinolaryngology, Jikei University School of Medicine, Tokyo, Japan

3-25-8 Nishi-shinbashi, Minato-ku, Tokyo 105-8461, Japan Phone: 81-3-3433-1111, ext 3601 Fax: 81-3-5400-1250 E-mail: [email protected] [email protected]

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Abstract

surgery for secondary acquired cholesteatoma (SAC).

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Objective: This study aimed to analyze the clinical features of patients who underwent

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Materials and methods: The subjects were 30 patients who underwent surgery for

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SAC in 30 ears. We investigated the age distribution, sex, tympanic membrane (TM) findings, temporal bone pneumatization, morphology of TM epidermis invasion, extent of cholesteatoma invasion, ossicular erosion, surgical methods and surgical results.

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Results: There were 10 males (33.3%) and 20 females (66.6%), with a mean age 54.9

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years. The TM perforation was medium-sized or larger in 27 ears (90%). Temporal bone

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pneumatization was poor or bad in 90% (18/20) of the evaluated ears. The cholesteatoma invaded from the malleus manubrium to the promontory in 23 ears

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(76.7%). There were no patients in whom the cholesteatoma invaded to the antrum or mastoid. The ossicles were affected in 19 ears (63.3%). Ossiculoplasty with a columella on the stapes was the most frequent procedure, performed for 16 ears (53.3%). There were no hearing results with a postoperative air-bone gap of more than 31 dB. Conclusions: Although SAC is rare, it is important for the clinician to keep this type of cholesteatoma in mind.

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1. Introduction

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Most cases of acquired middle ear cholesteatoma are considered to develop due to retraction or adhesion of the tympanic membrane (TM). However, there

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are some cases in which the TM epidermis invades to the medial surface of the

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TM from the margin of its perforation, without any retraction or adhesion. This is called secondary acquired cholesteatoma (SAC). To the best of our knowledge, few studies of SAC have been reported in the literature. Although SAC is quite

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rare, it is an actual clinical entity and clinicians must be aware of its existence.

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The objective of this study was to analyze the clinical features of patients who

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underwent surgery for SAC.

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2. Materials and methods The subjects were 30 patients with SAC in 30 ears, for which operations (primary surgery) were performed at the Jikei University Hospital, Tokyo, Japan, from July 1989 through December 2011. The patients were followed for at least 1 year after their last operation. The presence or absence of a history of otitis media was not taken into consideration. There are no definitive criteria for SAC. We used the following criteria to

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diagnose SAC in this study: (1) presence of perforation of the pars tensa, without

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any TM retraction or adhesion; (2) TM epidermis invading from the margin of the perforation to the medial surface of the TM; and (3) removal of the invaded TM

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epidermis requires a surgical procedure in the tympanic cavity.

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We analyzed the following features: the age distribution, sex, TM findings, temporal bone pneumatization, morphology of TM epidermis invasion, extent of the cholesteatoma invasion, ossicular erosion, surgical methods and surgical

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results.

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3. Results

3.1. Age distribution and sex

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There were 10 males (33.3%) and 20 females (66.6%), with a mean age of 54.9 years (range, 27–71 years), with the largest number being 50 to 59 years of age (Fig. 1). Twenty-two patients (73.3%) were 50 years or older.

3.2. Size of tympanic membrane perforation The TM perforation was assessed as total (almost no remaining, intact TM) in 10 of the 30 ears (33.3%), large (involving more than 3 tympanic quadrants) in 5

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(16.7%), medium (involving 2 tympanic quadrants) in 12 (40%) and small

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(involving a single tympanic quadrant) in 3 (10%).

Thus, the perforation was medium or larger in size in 90% (27/30 ears) of the

3.3. Preoperative diagnosis

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patients.

Based on the preoperative TM findings or CT findings, 19 of the 30 (63.3%)

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ears were diagnosed as cholesteatoma, and 11 (36.7%) were diagnosed as

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chronic otitis media or tympanosclerosis without cholesteatoma.

3.4. Preoperative findings of calcification

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Calcification surrounding the TM or the ossicles was found in 12 (40%) of the 30 ears. Seven of those 12 ears had been diagnosed as chronic otitis media or tympanosclerosis without cholesteatoma preoperatively.

3.5. Temporal bone pneumatization In this study, the degree of temporal bone pneumatization was classified as good (good development of air cells), poor (suppressed development of air cells),

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or bad (no development of air cells). The result of classification in 20 patients for

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whom data were available was good in 2 (10%), poor in 11 (55%) and bad in 7

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(35%).

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3.6. Morphology of TM epidermis invasion

Based on the site and pattern of invasion of TM epidermis, the morphology of the cholesteatomas was classified into two types. In Type 1, the TM epidermis

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has invaded from the malleus manubrium to the promontory. In Type 2, the TM

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epidermis has invaded from the edge of the perforation along the inner surface

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of the TM. Twenty-three of the 30 ears (76.7%) were classified as Type 1, and

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the remaining 7 as Type 2 (23.3%).

3.7. Extent of cholesteatoma invasion Based on our analysis of the surgical findings, we classified the extent of invasion by the cholesteatoma into three types: localized within the inner surface of the TM (the cholesteatoma invasion ends at the inner surface of the TM), extends to the mesotympanum, or reaches the attic. There were no ears in which the cholesteatoma invaded to the antrum or mastoid.

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In 17 (73.9%) of the 23 ears with Type 1 SAC , the cholesteatoma reached the

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attic. On the other hand, in 6 of the 7 (85.7%) Type 2 ears, the cholesteatoma did not invade past the mesotympanum. That is, it was localized within the inner

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surface of the TM in 3 ears and invaded to the mesotympanum in 3 ears. In the

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final (14.3%) Type 2 ear, the cholesteatoma reached the attic.

3.8. Ossicular erosion

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Table 2 shows the findings regarding ossicular erosion. The ossicles were

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affected in 19 (63.3%) of the 30 ears. In particular, there was some destruction

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of the incus in 18 ears (60%).

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3.9. Surgical methods

Table 3 shows the details of the surgical methods. Canal wall up tympanoplasty was performed on 4 (13.3%) ears, tympanoplasty with transcanal atticotomy on 15 (50%) ears and tympanoplasty without mastoidectomy on 11 (36.7%) ears. Planned staged tympanoplasty was performed on 1 ear. Five ears with cholesteatoma localized within the inner surface of the TM all underwent tympanoplasty without mastoidectomy.

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For 18 ears with cholesteatoma reaching to the attic, the most frequent

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surgical method was tympanoplasty with transcanal atticotomy, performed on 13 (72.2%) of those ears.

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We were able to treat most (i.e., 26 of the total 30; 86.7%) of these SAC cases

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by tympanoplasty without mastoidectomy or with transcanal atticotomy, since the cholesteatoma did not invade beyond the attic.

3.10. Ossiculoplasty

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Ossiculoplasty with a columella on the stapes was the most frequent

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procedure, performed for 16 (53.3%) ears (Table 4). Wullstein Type I method

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was applied for all 5 ears with cholesteatoma localized within the inner surface of

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the TM. Ossiculoplasty with a columella on the foot plate was performed for 7 ears because of destruction of the stapes’ superstructure.

3.11. Surgical results We examined the postoperative hearing of 19 patients whom we were able to follow for at least 1 year after surgery. Each patient underwent preoperative and postoperative audiologic evaluation. Four ABG frequencies, i.e., 0.5, 1, 2 and 4 kHz, were used to calculate pure tone averages. In Japan, 3 kHz is not

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commonly used; therefore, 4 kHz was substituted for 3 kHz in this study. As a

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result, the largest number of ears had a postoperative air-bone gap of 11-20 dB, i.e., 8 (42.1%) ears. There were no hearing results with a postoperative air-bone

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gap of 31 dB or more (Table 5).

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None of the 30 ears experienced recurrence of the cholesteatoma or

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re-perforation of the TM.

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4. Discussion

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Histological studies have reported migration of stratified squamous epithelium from the lateral surface of the TM through a perforation in the TM. Oktay et al.

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found migrated stratified squamous epithelium on the inner surface of the TM in

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11 of 29 human temporal bones (38%) [1]. Yamashita performed a histological study on 40 human temporal bones with TM perforations (including central and marginal perforations) and found that the mucocutaneous junction was located

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medial to the margin of the perforation in 11 ears (27.5%) [2]. Somers et al.

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found that histological localization of the muco-epithelial junction showed a

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medial position in 30%, with extensive middle ear invasion by stratified squamous epithelium in 7% [3]. They suggested that a margin of at least 1.5 mm

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should be removed if no clear infiltration is observed along the medial side of the TM. Although TM perforations are typical findings of chronic otitis media, these studies provide evidence that TM perforations are not always as benign as generally thought, and that the margins of the perforation can hide a cholesteatoma. TM findings give important Information for diagnosis of the middle ear disease, and TM perforations are one of the features of SAC above all. However, in cases

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with calcification surrounding the TM or ossicles, distinction of simple chronic

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otitis media with a central TM perforation and SAC is sometimes difficult only from TM findings. In this study, 7 of 12 ears with calcification surrounding the TM

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or ossicles had been preoperatively diagnosed as chronic otitis media or

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tympanosclerosis without cholesteatoma. Since preoperative diagnosis may be difficult in cases with calcification surrounding the TM or ossicles, careful preoperative inspection of the TM using an otomicroscope, otoendoscope, etc.,

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is important. And in order to diagnose SAC above all, it is important to inspect

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well the presence of surgical findings of continuous epithelial invasion from the

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perforation edge to the back side of the TM and accumulation of keratin debris using an otoendoscope, etc.

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In some cases of chronic otitis media the TM epidermis extends slightly onto the medial surface of the TM from the margin of its perforation, but development of cholesteatoma is rare in actual clinical settings [4]. Usually, if epidermis invades along the surface of a tissue and contacts mucosal epithelium, the invasion stops [5]. However, chronic inflammation leads to degeneration and deciduation of the middle ear mucosal epithelium, and we hypothesize that the epidermis invades in an attempt to cover the degenerated middle ear mucosal

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epithelium. Various cytokines induced by chronic inflammation and infection

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promote differentiation and proliferation of the epidermis [6-9]. The causes of SAC are degeneration and deciduation of the middle ear mucosal epithelium,

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and differentiation and proliferation of the epidermis. Moreover, chronic

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inflammation is thought to be strongly involved in the development of SAC. Wayoff et al. classified epidermal invasion into three types: (1) invasion from the malleus manubrium, (2) invasion from the incus long process to around the

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stapes and (3) invasion from the lateral surface of the TM through a perforation

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in its inner surface [9]. In this study, analysis of the site and pattern of invasion of

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TM epidermis showed that Type 1 (76.7%) was more common than Type 2 (23.3%) in the patients. Our Type 1 corresponds to the “epidermis invasion from

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the malleus manubrium” type described by Wayoff et al [9]. Yamamoto et al. suggested that the common area of the epithelial invasion of SAC was on the superior part of the TM perforation around the tip of the malleus handle. Our results that Type 1 was more common than Type 2 support their hypothesis [10]. This was probably because the TM around the malleus manubrium was the origin of the TM migration, and this region has many capillaries and a rich blood supply [11]. Therefore, the epidermis was activated, and then the TM epidermis

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easily invaded from the perforation margin around the malleus manubrium.

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Analysis of temporal bone pneumatization found that it was poor or bad in most cases (18 of 20). This was probably because the time-course of chronic

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otitis media with a central perforation of the TM was very long, and chronic

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inflammation persisted. That might have resulted in suppressed development of temporal bone air cells. The long course of chronic otitis media is a characteristic of SAC, and there were thus few young patients. It has been reported that

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establishment of SAC requires long periods of time, because the average age of

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SAC patients was higher than that of patients with other types of acquired

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cholesteatoma [10]. Also our results support the concept that long-term chronic inflammation is involved in generation of SAC.

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Establishment of SAC requires degeneration and deciduation of the middle ear mucosal epithelium, and differentiation and proliferation of the epidermis, which are promoted by various cytokines that are induced by long-term chronic inflammation and infection. It can be surmised that SAC develops when there is convergence of these various conditions due to chronic inflammation. However, it is rare for these conditions to be met, and thus the incidence of SAC is considered to be low.

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Our analysis of the extent of invasion of the cholesteatoma showed that it

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reached the attic in most cases of Type 1 morphology. On the other hand, in all but one of the Type 2 morphology cases the cholesteatoma did not invade

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beyond the mesotympanum. For all 5 ears in which the cholesteatoma was

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localized within the inner surface of the TM, we performed tympanoplasty without mastoidectomy and applied the Wullstein type I method. In all 30 ears, the cholesteatoma did not invade beyond the attic. There were thus no ears in

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which the cholesteatoma invaded to the antrum or mastoid, and a characteristic

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of SAC is that the invasion is not much deeper than in other types of acquired

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cholesteatoma. Therefore, in most patients the cholesteatoma matrix can be eliminated by tympanoplasty without mastoidectomy or by transcanal atticotomy,

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since it usually remains in the mesotympanum or attic. In most cases with invasion of the cholesteatoma to the mesotympanum or attic, the stratified squamous epithelium invades to the promontory via the tendon of the tensor tympani muscle. Therefore, careful inspection and sufficient surgical elimination of cholesteatoma around the tendon using various instruments, such as an otoendoscope, are required so that no cholesteatoma is left.

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In this study, there were no cases of recurrence of the cholesteatoma or

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re-perforation of the TM and no hearing results with a postoperative air-bone gap of 31 dB or more. Furthermore, a characteristic of SAC is thought to be that the

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invasion is not much deeper than in other types of acquired cholesteatoma. In

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selecting an appropriate surgical method and procedure, postoperative progress of SAC is thought to be good. As mentioned above, Somers et al. suggested that a margin of at least 1.5 mm should be removed along the medial side of the TM.

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Especially, since preoperative diagnosis may be difficult in cases with

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calcification surrounding the TM or ossicles, a sufficient margin should be

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removed in such cases with calcification. In order to prevent the recurrence and acquire postoperative good hearing ability, it is much important to select the

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appropriate surgical method and ossiculoplasty according to each case.

5. Conclusion We analyzed the clinical features of 30 patients with SAC who were operated on in our department. Careful inspection is required because a cholesteatoma may be hidden even when it appears that there is only simple chronic otitis media with a central perforation of the TM. Although SAC is rare, it is important

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for the clinician to keep this type of cholesteatoma in mind. Although the

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pathogenesis of this type of cholesteatoma remains unclear, several possible theories were discussed. We hope to perform careful examination and analysis

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of a larger number of cases.

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Figure and Table Legends

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Fig. 1. Age distribution and sex.

Table 1. Extent of cholesteatoma invasion.

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Twenty-two of the 30 patients (73.3%) were 50 years or older.

antrum or mastoid. Table 2. Ossicular erosion.

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There were no patients (ears) in whom the cholesteatoma invaded to the

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The ossicles were affected in 19 (63.3%) of the 30 ears.

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Table 3. Surgical methods.

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Table 4. Ossiculoplasty.

Ossiculoplasty with a columella on the stapes was the most frequent

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procedure, performed for 16 ears (53.3%). Table 5. Hearing results. There were no hearing results of 31 dB or more for the postoperative air-bone gap.

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