Short-Term Hearing Prognosis of Ossiculoplasty in Pars Flaccida Cholesteatoma Using the EAONO/JOS Staging System

Abstract

Objectives:
The aim of the present study was to investigate the prognostic factors for short-term hearing outcomes of ossiculoplasty for primary pars flaccida cholesteatoma according to the European Academy of Otology and Neurotology/Japanese Otological Society (EAONO/JOS) and 2015 JOS staging systems.

Materials and methods:
A total of 34 patients with primary pars flaccida cholesteatoma who underwent one-stage tympanomastoidectomy with partial ossicular reconstruction using double cartilage block were included in the study. The postoperative pure-tone average air-bone gap (PTA-ABG) was calculated, and two criteria of successful hearing outcomes were defined as ≤10 and ≤20 dB. Patients were classified according to the EAONO/JOS and 2015 JOS staging systems. Cochran-Armitage test was used to statistically analyze staging, and Fisher's exact test was used to analyze other factors.

Results:
Successful hearing outcome with postoperative PTA-ABG ≤10 and ≤20 dB occurred in 23.5% and 55.9% of cases, respectively. When postoperative PTA-ABG ≤20 dB was defined as successful, the success rate significantly decreased with increase in EAONO/JOS stage, and S0 pathological status of the stapes (no involvement) was a significantly favorable predictive factor. When postoperative PTA-ABG ≤10 dB was regarded as successful, the significantly favorable predictive factors were S0 pathological status of the stapes and development of mastoid cells with MC2-3 (better developed cells).

Conclusion:
Favorable prognostic factors for hearing outcomes of tympanomastoidectomy with partial ossicular reconstruction for primary pars flaccida cholesteatoma were low stage following the EAONO/JOS staging system and no stapes involvement and better development of mastoid cells following the 2015 JOS staging system.

Full text

J Int Adv Otol 2019; 15(1): 2-7 • DOI: 10.5152/iao.2019.5983
Original Article
INTRODUCTION
Pars accida cholesteatoma (attic cholesteatoma), which originates in a pars accida retraction pocket, is a non-neoplastic cystic
lesion formed by keratinizing squamous epithelium and keratin debris. It can gradually expand into the middle ear and cause
complications by the erosion of the nearby bony structures. There has been no viable non-surgical therapy developed [1]. Thus,
tympanoplasty is performed to remove pathological lesions and maintain or improve hearing.
In 2015, the Japan Otological Society (JOS) has proposed staging and classication criteria for middle ear cholesteatoma to pro-
vide a basis for meaningful exchange of information pertaining to cholesteatoma treatment [2, 3]. In 2017, the European Academy
of Otology and Neuro-otology (EAONO) and the JOS have collaborated and published joint consensus statements regarding the
denition, classication, and staging of middle ear cholesteatoma [4].
Short-Term Hearing Prognosis of Ossiculoplasty in
Pars Flaccida Cholesteatoma Using the EAONO/JOS
Staging System
OBJECTIVES: The aim of the present study was to investigate the prognostic factors for short-term hearing outcomes of ossiculoplasty for primary
pars flaccida cholesteatoma according to the European Academy of Otology and Neurotology/Japanese Otological Society (EAONO/JOS) and
2015 JOS staging systems.
MATERIALS and METHODS: A total of 34 patients with primary pars flaccida cholesteatoma who underwent one-stage tympanomastoidectomy
with partial ossicular reconstruction using double cartilage block were included in the study. The postoperative pure-tone average air–bone gap
(PTA-ABG) was calculated, and two criteria of successful hearing outcomes were defined as ≤10 and ≤20 dB. Patients were classified according to
the EAONO/JOS and 2015 JOS staging systems. Cochran–Armitage test was used to statistically analyze staging, and Fisher’s exact test was used
to analyze other factors.
RESULTS: Successful hearing outcome with postoperative PTA-ABG ≤10 and ≤20 dB occurred in 23.5% and 55.9% of cases, respectively. When
postoperative PTA-ABG ≤20 dB was defined as successful, the success rate significantly decreased with increase in EAONO/JOS stage, and S0
pathological status of the stapes (no involvement) was a significantly favorable predictive factor. When postoperative PTA-ABG ≤10 dB was re-
garded as successful, the significantly favorable predictive factors were S0 pathological status of the stapes and development of mastoid cells
with MC2–3 (better developed cells).
CONCLUSION: Favorable prognostic factors for hearing outcomes of tympanomastoidectomy with partial ossicular reconstruction for primary
pars flaccida cholesteatoma were low stage following the EAONO/JOS staging system and no stapes involvement and better development of
mastoid cells following the 2015 JOS staging system.
KEYWORDS: Cholesteatoma, tympanoplasty, prognosis, hearing
Atsushi Fukuda , Shinya Morita , Yuji Nakamaru , Kimiko Hoshino , Keishi Fujiwara ,
Akihiro Homma
Department of Otolaryngology-Head and Neck Surgery, Hokkaido University, School of Medicine and Graduate School of Medicine, Hokkaido,
Japan
Corresponding Author: Fukuda Atsushi E-mail: atsushi.fukuda@huhp.hokudai.ac.jp
Submitted: 27.08.2019 • Revision Received: 07.03.2019 • Accepted: 15.03.2019
©Copyright 2019 by The European Academy of Otology and Neurotology and The Politzer Society - Available online at www.advancedotology.org
ORCID IDs of the authors: A.F. 0000-0001-9204-0007; S.M. 0000-0002-2429-9751; Y.N. 0000-0002-9045-4617; K.H. 0000-0001-8582-7936; K.F. 0000-
0002-4770-3227; A.H. 0000-0003-1488-0646.
Cite this article as: Fukuda A, Morita S, Nakamaru Y, Hoshino K, Fujiwara K, Homma A. Short-Term Hearing Prognosis of Ossiculoplasty in Pars Flac-
cida Cholesteatoma Using the EAONO/JOS Staging System. J Int Adv Otol 2019; 15(1): 2-7.
This study was presented at the “9th EAONO Instructional Workshop”, “20th-23th of June 2018”, “Copenhagen, Denmark”.
2

Although various factors aecting the hearing outcomes of tympa-
noplasty have been reported [5-17], to the best of our knowledge, no
study has investigated the favorable prognostic factors for hearing
outcomes of ossiculoplasty for middle ear cholesteatoma based on
standardized staging and classication criteria. The aim of the present
study was to investigate the prognostic factors for hearing outcomes
of one-stage tympanoplasty for primary pars accida cholesteatoma
according to the EAONO/JOS staging system [4], the 2015 JOS staging
system [2], and the factors listed in previous reports [5, 6, 8, 10-17].
MATERIALS AND METHODS
Patients
This was a retrospective study. A total of 34 consecutive patients who
underwent ossiculoplasty for primary pars accida cholesteatoma at
a university hospital between April 2013 and July 2017 were included
in the study and were followed up for >1 year. All patients underwent
one-stage tympanoplasty with mastoidectomy. Cholesteatoma was
diagnosed according to the EAONO/JOS joint consensus statements
on the denition, classication, and staging of middle ear choleste-
atoma [4]. Inclusion criteria only included patients with cartilage os-
siculoplasty with partial ossicular reconstruction in the presence of
stapes superstructure. The present study was approved by the Insti-
tutional Review Board of our university hospital for clinical research
(IRB no. 017-0375) according to the tenets of the Declaration of Hel-
sinki. Informed consent was not required for this retrospective study.
Surgical Procedure
All procedures were performed under general anesthesia. A postau-
ricular incision was made, and the cholesteatoma was microscopi-
cally removed using a canal wall up (CWU) or canal wall down (CWD)
technique. The technique was selected depending on the tegmen
height, degree of mastoid cell development, and presence or ab-
sence of tegmen destruction. An endoscope was used to examine if
there was any residual lesion when the cholesteatoma involved di-
cult access sites, such as the supratubal recess and sinus tympani. The
incus and the malleus head were then removed. A piece of cavum
conchae cartilage was harvested and used for ossicular reconstruc-
tion. Two small pieces of cartilage were prepared, and a shallow ac-
etabulum was created to receive the stapes capitulum on one of the
cartilage pieces. A double cartilage block was interposed between
the head of the stapes and the tympanic membrane. When the chor-
da tympani nerve was preserved, it was positioned on the double
cartilage block to stabilize it. A large meatoplasty was performed by
removing a segment of the conchal cartilage, and an inferiorly pedi-
cled, periosteal-pericranial ap [18] was used to partially obliterate the
mastoid cavity in all patients who underwent CWD tympanoplasty.
Staging and Classication Criteria
The patients were classied according to the EAONO/JOS staging
system. The extension of cholesteatoma in each ear was surgically
conrmed and scored according to middle ear involvement using
the STAM system: S1 (supratubal recess), S2 (sinus tympani), T (tym-
panic cavity), A (attic), and M (mastoid) (Figure 1) [4]. The staging sys-
tem for pars accida cholesteatoma was as follows: I (cholesteatoma
localized in the attic), II (cholesteatoma involving two or more sites),
III (cholesteatoma with extracranial complications), and IV (choles-
teatoma with intracranial complications) (Table 1) [4]. Mastoid cell
development was assessed by preoperative computed tomography
and classied into one of the four degrees following the 2015 JOS
staging system: MC0 (almost no cell growth), MC1 (cellular structures
only around the mastoid antrum), MC2 (well-developed cellular
structures), and MC3 (cellular structures extending to the peri-laby-
rinthine area) (Figure 2) [2]. The pathological status of the stapes was
Table 1. The EAONO/JOS staging system for pars accida cholesteatoma
(attic cholesteatoma) (Adapted from Yung et al. 2017 [4])
Stage I Cholesteatoma localized in the attic
Stage II Cholesteatoma involving two or more sites
Stage III Cholesteatoma with extracranial complications or pathologic
conditions including
Facial palsy,
Labyrinthine fistula: with conditions at risk of membranous
labyrinth,
Labyrinthitis,
Postauricular abscess or fistula,
Zygomatic abscess,
Neck abscess,
Canal wall destruction: more than half the length of the bony ear
canal,
Destruction of the tegmen: with a defect that requires surgica
repair, and
Adhesive otitis: total adhesion of the pars tensa.
Stage IV Cholesteatoma with intracranial complications including
Purulent meningitis,
Epidural abscess,
Subdural abscess,
Brain abscess,
Sinus thrombosis, and
Brain herniation into the mastoid cavity.
Figure 1. Schematic of divisions of the middle ear space using STAM system: S1,
supratubal recess; S2, the sinus tympani; T, tympanic cavity; A, attic; M, mastoid
(Adapted from Yung et al. [4] 2017).
3
Fukuda et al. Prognosis of Ossiculoplasty in Cholesteatoma

intraoperatively evaluated and classied into two statuses following
the 2015 JOS staging system: S0 (no stapes involvement) and S1 (su-
perstructure surrounded by cholesteatoma and/or granulation) (Fig-
ure 3) [2]. The condition of the malleus handle was dened as absent
when the malleus handle was eroded due to a lesion or when it was
purposefully removed. The tympanic cavity mucosa (e.g., edematous
or adhesive) was intraoperatively identied as either normal or dis-
eased.
Hearing Outcome
Hearing outcome was calculated according to the guidelines of the
Committee on Hearing and Equilibrium of the American Academy of
Otolaryngology-Head and Neck Surgery [19]. Pure-tone air-conduc-
tion and bone-conduction thresholds were obtained with thresholds
at 0.5, 1, 2, and 3 kHz, which were used to calculate the pure-tone av-
erage air–bone gap (PTA-ABG). When 3 kHz was not tested, the mean
thresholds at 2 and 4 kHz were used instead, and a four-frequency
(0.5, 1, 2, and 3 kHz) PTA-ABG was calculated. Audiograms at ≥1 year
but <2 years after surgery were used for determining postoperative
short-term hearing results. The two criteria of successful hearing out-
comes were dened as PTA-ABG ≤10 and ≤20 dB.
Statistical Analysis
JMP pro 14 (SAS Institute, Inc., Cary, NC, USA) was used for statistical
analysis. The prognostic factors for hearing outcomes were analyzed.
The prognostic factors were patient age (<60/≥60 years), staging (I-
Figure 2. Axial CT images representing degrees of mastoid cell development
(MC0-MC3) (Adapted from Tono et al. [2] 2017).
Figure 3. Schematic of criteria for pathological status of the stapes (S0 and S1)
(Adapted from Tono et al. [2] 2017).
Table 2. Demographic and clinical data of the patients
Characteristics No. (%)
Sex
Male 18 (52.9)
Female 16 (47.1)
Age, years
median (range) 61.5 (16–87)
<60 15 (44.1)
≥60 19 (55.9)
Staging
Stage I 5 (14.7)
Stage II 16 (47.1)
Stage III 13 (38.2)
Labyrinthine fistula 3 (8.8)
Destruction of the tegmen 10 (29.4)
Adhesive otitis 1 (2.9)
Stage IV 0 (0)
S1 involvement
+ 1 (2.9)
− 33 (97.1)
S2 involvement
+ 3 (8.8)
− 31 (91.2)
T involvement
+ 5 (14.7)
− 29 (85.3)
M involvement
+ 26 (76.5)
− 8 (23.5)
Development of mastoid cells
MC0-1 27 (79.4)
MC2-3 7 (20.6)
Pathological status of the stapes
S0 18 (52.9)
S1 16 (47.1)
Surgical procedure
CWU 14 (41.2)
CWD 20 (58.8)
Malleus handle
Present 30 (88.2)
Absent 4 (11.8)
Cholda tympani nerve
Present 23 (67.6)
Absent 11 (32.4)
Middle ear mucosa
Normal 30 (88.2)
Diseased 4 (11.8)
CWU: canal wall up; CWD: canal wall down.
4
J Int Adv Otol 2019; 15(1): 2-7

IV), S1 involvement, S2 involvement, T involvement, M involvement,
development of mastoid cells (MC0-1/MC2-3), pathological status of
the stapes (S0/S1), surgical procedure (CWU/CWD), malleus handle
(present/absent), chorda tympani nerve (present/absent), and mid-
dle ear mucosa (normal/diseased). Cochran–Armitage test was used
for statistical analysis of staging, and Fisher’s exact test was used for
statistical analysis of other factors. A p<0.05 was considered statisti-
cally signicant.
RESULTS
A total of 34 patients were enrolled in the study. The study included
18 male patients. The median age of the patients was 61.5 (16-87)
years. The mean follow-up period was 41 (12-63) months. Table 2
shows the demographic and clinical data of the patients. There were
no patients with stage IV pars accida cholesteatoma. Table 3 shows
the hearing outcomes of all patients. Successful hearing outcomes
with postoperative PTA-ABG ≤10 and ≤20 dB were observed in 23.5%
and 55.9% of the cases, respectively. One out of the 34 patients had
revision surgery for a recurrent cholesteatoma 3 years after undergo-
ing CWU tympanoplasty. There were no patients who experienced
postoperative complications, such as local ap necrosis, cavity prob-
lems, facial nerve paralysis, meningitis, or brain abscess.
Figure 4 and Table 4 show the analysis of the prognostic factors for
hearing outcomes. When postoperative PTA-ABG ≤20 dB was used to
dene successful hearing outcomes, the successful hearing improve-
ment rate signicantly decreased with increase in the EAONO/JOS
stage (p=0.0249), and the S0 pathological status of the stapes (no
stapes involvement) was a signicantly favorable predictive factor
(p=0.0142). When postoperative PTA-ABG ≤10 dB was used to dene
successful hearing outcomes, the signicantly favorable predictive
factors were S0 pathological status of the stapes (p=0.0425) and de-
velopment of mastoid cells with MC2-3 (better developed mastoid
cells) (p=0.0374). The cholesteatoma extent according to the STAM
system, surgical procedure, presence of the malleus handle and chor-
da tympani nerve, and middle ear mucosal status were not signicant
predictors for any of the criterion of successful hearing outcomes.
DISCUSSION
Many studies have investigated the prognostic factors for a successful
ossiculoplasty. The favorable prognostic factors aecting outcomes
in ossicular chain reconstruction are a low level of otorrhea [6, 16], the
presence of malleus handle [6, 8, 12, 13, 15-17], the presence of stapes super-
Table 4. Analysis of the prognostic factors of hearing outcomes
p*
Postoperative PTA-ABG
Factors Contrast ≤10 dB vs >10 ≤20 dB vs >20
Age, years <60 N.S. N.S.
≥60
S1 involvement + N.S. N.S.
−
S2 involvement + N.S. N.S.
−
T involvement + N.S. N.S.
−
M involvement + N.S. N.S.
−
Development of MC0–1 0.0374 N.S.
mastoid cells MC2–3
Pathological status S0 0.0425 0.0142
of the stapes S1
Surgical procedure CWU N.S. N.S.
CWD
Malleus handle Present N.S. N.S.
Absent
Chorda tympani nerve Present N.S. N.S.
Absent
Mucosa Normal N.S. N.S.
Diseased
PTA-ABG: pure-tone average air-bone gap; N.S.: not significant; CWU: canal wall up;
CWD: canal wall down.
* Fischer’s exact test
Figure 4. The rate of successful hearing improvement signicantly decreased
with increase in EAONO/JOS statege (p=0.0249).
Table 3. Hearing outcomes
Postoperative data
Mean air-bone gap (SD) 19.2 (10.4) dB
Air-bone gap
0-10 dB 8 (23.5 %)
10-20 dB 11 (32.4 %)
20-30 dB 11 (32.4 %)
>30 4 (11.8 %)
SD: standard deviation.
5
Fukuda et al. Prognosis of Ossiculoplasty in Cholesteatoma

structure [7, 9, 12, 14, 17], normal stapes mobility [12], the presence of chorda
tympani nerve [10], normal middle ear mucosal status [12-14, 16], intact
canal wall tympanomastoidectomy (CWU) [5, 10, 11, 16], primary surgery
[9-11, 16, 17], and local anesthesia [12]. Although various factors that aect
postoperative hearing outcomes have been reported, they are not al-
ways signicant, and the results have been controversial. In addition,
many studies have adapted many pathological conditions and used
various methods of ossicular chain reconstruction in their studies [6-
17], and only a few studies have been limited to cholesteatoma [5].
Hearing outcomes of tympanoplasty for middle ear cholesteatoma
are of interest to otologists worldwide. However, there are no com-
mon standards for discussion of the postoperative results. The EAO-
NO/JOS and JOS 2015 staging systems for middle ear cholesteatoma
have been recently published [2, 4], Hence, postoperative hearing re-
sults were studied based on these staging systems. Our research was
limited to primary pars accida cholesteatoma. Furthermore, the sur-
gical procedure was limited to one-stage tympanomastoidectomy
with partial ossicular reconstruction using the double cartilage block
in the presence of stapes superstructure to reduce confounders.
In the present study, the postoperative PTA-ABG was 18.0 dB, and
successful hearing outcome with a postoperative PTA-ABG ≤20 dB
occurred in 55.9% of the cases. According to the literature, the clo-
sure of ABG to within 20 dB ranges from 50% to 85.2% using the
double cartilage block [20-23]. Our results are in accordance with these
studies, although the latter included various pathological conditions
other than primary pars accida cholesteatoma, and a direct com-
parison is dicult.
In our study, the rate of successful hearing improvement signicantly
decreased with increase in EAONO/JOS stage, and S0 pathological
status of the stapes (no involvement) was a signicantly favorable
predictive factor when postoperative PTA-ABG ≤20 dB was consid-
ered a successful hearing outcome. When postoperative PTA-ABG
≤10 dB was used to dene successful hearing outcome (excellent
results), both S0 pathological status of the stapes and development
of mastoid cells (MC2-3, better development) were signicantly fa-
vorable predictive factors. Cholesteatoma and granulation can cause
the deterioration of mobility in stapes with S1 pathological status
(superstructure surrounded by cholesteatoma and/or granulation).
Therefore, the hearing results of cases with S0 might be better than
those with S1 status. Mishiro et al. [12] reported that normal stapes
mobility is a signicantly favorable predictor of ossiculoplasty, and
their results are consistent with those reported in the present study.
Some authors have demonstrated the important role of postopera-
tive aeration in the middle ear in achieving better hearing outcomes
of tympanoplasty [23-25]. Better developed mastoid cells, which indi-
cate good Eustachian tube function, may contribute to aerated tym-
panomastoid cavities postoperatively. Hence, the hearing outcome
of cases with MC2–3 might be better than those with MC0-1.
There was a clear correlation between the rate of successful hearing
improvement and EAONO/JOS stage, indicating that the EAONO/JOS
stage reects the hearing prognosis after partial ossiculoplasty for
primary pars accida cholesteatoma. On the other hand, no correla-
tion was found between hearing outcome and the involvement of
particular sites using the STAM system. Surgical procedure, presence
of the malleus handle and chorda tympani nerve, and middle ear mu-
cosal status were also not signicant predictors of successful hearing
in our study. The small sample size and/or some confounders may
have been the cause of these factors not being signicant.
Our study had some limitations. Since the present study was ret-
rospective, only short-term hearing outcomes were investigated.
Moreover, only univariate analysis was performed. Multivariate anal-
ysis could not be performed because of the small number of samples.
Therefore, confounding factors could not be avoided. Further inves-
tigations are required with multivariate analysis of a large number of
samples in a prospective survey according to a standardized basis for
evaluation, such as the EAONO/JOS staging system, to reveal inde-
pendent signicant prognostic factors of ossiculoplasty for middle
ear cholesteatoma.
CONCLUSION
No stapes involvement and low EAONO/JOS stage were the favor-
able prognostic factors for hearing outcomes of ossiculoplasty with
partial ossicular reconstruction for primary pars accida cholestea-
toma. In particular, there may be a strong association between the
accomplishment of excellent hearing results and development of
mastoid cells. Therefore, the EAONO/JOS staging and the criteria
for evaluation of the pathological status of stapes and the degree of
mastoid cell development in the 2015 JOS staging systems may be
useful for predicting the prognosis of hearing outcomes of partial os-
siculoplasty for primary pars accida cholesteatoma.
Ethics Committee Approval: Ethics committee approval was received for this
study from the Institutional Review Board of Hokkaido University Hospital for
clinical research (IRB no. 017-0375) according to the tenets of the Declaration
of Helsinki.
Informed Consent: Informed consent is not necessary due to the retrospec-
tive nature of this study.
Peer-review: Externally peer-reviewed.
Author Contributions: Concept – A.F., S.M., Y.N., A.H.; Design - A.F., S.M., Y.N.,
A.H.; Supervision - Y.N., A.H.; Resource - A.H.; Materials - A.F.; Data Collection
and/or Processing - K.H.; Analysis and/or Interpretation - K.F.; Literature Search
- A.F.; Writing - A.F., K.H., K.F.; Critical Reviews - A.H.
Acknowledgements: The authors thank Enago (www.enago.jp) for the En-
glish language review.
Conict of Interest: The authors have no conict of interest to declare.
Financial Disclosure: The authors declared that this study has received no
nancial support.
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Fukuda et al. Prognosis of Ossiculoplasty in Cholesteatoma