Muigg2019 PDF
Muigg2019 PDF
Muigg2019 PDF
https://doi.org/10.1007/s00405-019-05737-6
OTOLOGY
Abstract
Purpose To determine the 2-year outcome of health-related quality of life (HRQoL) in adults who received a cochlear implant
(CI) for single-sided deafness (SSD).
Methods Twenty adults (mean age at implantation: 47 ± 11 years) with SSD (PTA worse ear: 113 dB HL, PTA better ear:
14 dB HL) were administered the Nijmegen Cochlear Implant Questionnaire (NCIQ), and the Health Utility Index 3 (HUI
3). Questionnaire administration occurred before cochlear implantation and 3, 6, 12, and 24 months after implant activation.
Results Of the 20 patients, 2 discontinued CI use within the observation period due to poor benefit. The NCIQ total score
of the sample increased significantly over time (p = 0.003). The largest increase occurred within the first 3 months of CI
use. Also, the HUI 3 multi-attribute utility score increased significantly (p = 0.03). The post-treatment increase of this score
(+ 0.11 points) indicated that the gain in HRQoL was clinically relevant. Patients with a duration of deafness > 10 years had
in all measures an equal HRQoL improvement than had patients with a duration of deafness < 10 years.
Conclusion Cochlear implantation led to significant improvement of hearing-specific and generic HRQoL in our patients.
The improvement was seen after 3 or 6 months but did not increase further at later intervals. Patients with long-lasting SSD
may be at higher risk of discontinuing CI use. However, if they adapt to the CI, they can experience an equal increase of
HRQoL as patients with a short duration of SSD.
Keywords Single-sided deafness · Cochlear implant · Health-related quality of life · Duration of deafness
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speech-in-noise tests and sound localization, when compared c. How rapidly does the improvement in HRQoL occur
to the unaided situation or to rehabilitation options such as after implantation with CI?
CROS and bone conduction devices [9–16]. The second
advantage of cochlear implantation in SSD is that it may The outcome of cochlear implantation in SSD is not only
reduce tinnitus severity and tinnitus distress [11, 14, 17–19]. of clinical interest, but also of economic interest. The cost-
Third, cochlear implantation in SSD also has positive effects effectiveness of CI in SSD is still a difficult topic to assess
on HRQoL. A number of studies have reported significant for authorities due to limited research and lack of utility
improvement of HRQoL in SSD patients after cochlear data published from this population. For this purpose, we
implantation. For instance, Härkönen et al. [20] adminis- decided to address the above questions with two different
tered the Glasgow Benefit Inventory (GBI) to seven SSD instruments: with an instrument that measures disease-spe-
patients before cochlear implantation and 6 months after CI cific HRQoL and an instrument that provides generic utility-
activation. They found that the GBI total score significantly based measures of HRQoL. Generic utility-based measures
increased between the two time-points. Similarly, Dillon have the potential to influence resource allocation decisions
et al. [21] administered the Abbreviated Profile of Hearing in health care policy making when considered in an eco-
Aid Benefit (APHAB), the Speech, Spatial and Qualities of nomic evaluation [28].
Hearing Scale (SSQ), and the Tinnitus Handicap Inventory
(THI) to 20 patients with SSD who received a CI. After
12 months of CI use, significant improvement was found
with all three questionnaires; in some cases, the improve- Method
ment was seen already at 1 month after CI activation. Finke
et al. [22] interviewed 19 SSD patients with a semi-struc- Study design
tured questionnaire to explore their experiences after coch-
lear implantation. Analysis of patient responses indicated The study was conducted as a retrospective analysis of ques-
that a major part of the sample had experienced a substantial tionnaire data obtained routinely within the CI program
change of HRQoL after CI activation. The patients were implemented at the University Clinic in Innsbruck and at
also administered the International Outcome Inventory for the University Clinic in Würzburg. Approval from the local
Hearing Aids (IOI-HA), and it was found that the scores of institutional Ethics Committees for conducting the study
the IOI-HA subscale “HRQoL” had significantly risen after was received. Within the CI program, two different HRQoL
the treatment. Positive effects of cochlear implantation on questionnaires (see below for description) were adminis-
HRQoL of SSD patients were also reported from Rösli et al. tered to the patients at the following five time-points: before
[23], Skarzinsky et al. [24] and Häußler et al. [25]. cochlear implantation and 3, 6, 12 and 24 months after CI
Mixed results, on the other side, were reported by Louza activation.
et al. [26]. These authors administered 4 questionnaires to
a sample of 10 SSD patients: the SSQ, the APHAB, the
Nijmegen Cochlear Implant Questionnaire (NCIQ) and the Sample
EuroQoL-5-Dimensions-3-Levels (EQ-5D-3L). The latter
is a tool for assessment of generic HRQoL, while the NCIQ The study sample consisted of 20 adults (12 males, 8
is a tool for assessment of hearing-specific HRQoL in CI females) who received a CI for unilateral hearing loss in
patients [27]. When the data were analysed 1 year after coch- 1 of the above named clinics. The patients were implanted
lear implantation, none of these four measures indicated a between September 2015 and December 2016 (study onset),
significant improvement of the sample’s total score. Only and were followed over a period of 2 years for data collec-
a few subscales of the NCIQ and the SSQ showed a sig- tion. Only SSD patients were included in the study who met
nificant increase. Based on these findings, the authors con- the following criteria:
cluded that not all SSD patients might benefit from cochlear
implantation. • aged 18 years or older,
The goal of our study was to assess long-term outcome of • for the first time implanted with a CI (no previous CI),
HRQoL in patients who received a CI for SSD. In particular, • hearing in the worse (implanted) ear: > 80 dB HL (i.e., at
our study addressed the following questions: least severe hearing loss, measured as pure-tone average
[PTA] over frequencies 0.5, 1, 2 and 4 kHz),
a. What is the outcome of HRQoL in SSD patients after • hearing in the better ear: ≤ 30 dB HL PTA (i.e., at worst
2 years of CI use? mild hearing loss),
b. How does HRQoL in SSD develop over time after
implantation with CI?
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• adequate speech–language skills (i.e., capable of follow- considered to cover the full range of abilities/disabilities rel-
ing all oral and written communication at clinical exami- evant to the human health status: Vision, Hearing, Speech,
nations and counselling without the help of others). Ambulation (i.e. mobility), Dexterity, Emotion, Cognition,
and Pain. The HUI 3 scoring system provides utility scores
The demographic and clinical data of the sample, includ- on a generic scale between values 1.00 (= perfect health)
ing causes for the deafness, are shown in Table 1. Of the 20 and 0.00 (= dead). Utility scores can be computed for each
patients, 11 had a duration of deafness longer than 10 years, of the eight attributes (single-attribute utility score [SAUS])
and 9 had a duration of deafness less than 10 years. These and for the general health-state (multi-attribute utility score
patients were assigned to two groups: long-duration SSD [MAUS]). Further information about the HUI 3 is summa-
and short-/medium-duration SSD, and the two groups were rized and reviewed in Horsman et al. [29].
considered separately in the statistical analysis. All the
patients were implanted with a MED-EL SYNCHRONY CI Statistical analysis
with a MEDEL electrode (either FLEX28 or FLEXSOFT),
and received a MED-EL SONNET speech processor. The NCIQ total score, SAUS and MAUS were each entered
in a repeated-measures ANOVA which included two fac-
Questionnaires tors: “Change over Time” (with the five measurement inter-
vals as levels) and “Duration of Deafness” (with two levels:
Two questionnaires were used to assess HRQoL in the sam- “more than 10 years” and “less than 10 years”). Sphericity
ple: the Nijmegen Cochlear Implant Questionnaire (NCIQ) assumption was tested with the Mauchly test and, if violated,
and the Health Utility Index 3 (HUI 3). Greenhouser–Geisser correction was applied to degrees of
The NCIQ [27] has been developed for assessment of freedom. Alpha-error level was set at 5%. Post hoc com-
hearing-specific HRQoL in CI patients. It consists of 60 parisons between the measurement intervals were performed
items covering 6 domains of hearing-specific HRQoL: with the paired-sample t test at a Bonferroni-adjusted alpha-
Basic Sound Perception (BSP), Advanced Sound Perception error level (α’ = 5% / 10 = 0.5%).
(ASP), Speech Production (SP), Self-Esteem (SE), Social Apart from significance, changes of the SAUS and the
interaction (SI), and Activity (Act). Responses to the items MAUS were also interpreted in view of their clinical rel-
are given on a five-step scale with the values 0/25/50/75/100. evance. It has been proposed to consider a difference of 0.05
Internal consistency (Cronbach’s alpha) of the six domains points or more between mean utility scores as clinically
has been reported in the range 0.73 < α < 0.89; retest reli- important [28]. This proposal has been based on the results
ability has been reported in the range 0.64 < r < 0.85 [26]. of studies, which compared the mean scores of patients with
The HUI 3 [29, 30] is a questionnaire for measuring certain clinical conditions (e.g. schizophrenia) to sex- and
generic and attribute-specific HRQoL and producing util- ethnicity-adjusted HUI population norms. Thereby, differ-
ity scores. It includes eight attributes (domains) which are ences as low as 0.03–0.05 points were found to indicate a
significant difference [29–32].
Table 1 Demographic and clinical data of SSD patients
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1.10 90
1.00 80
0.90 70
0.80 60
0.70 50
0.60 40
0.50
30
20
0.40
10
0.30
0
0.20
pre-operave 3 months 6 months 12 months 24 months
0.10
short/medium duraon of SSD long duraon of SSD
0.00
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that cochlear implantation leads to a quick improvement of The only study with divergent findings is the one of
hearing-specific HRQoL which, however, does not further Louza et al. [26]. This study also used the NCIQ for HRQoL
increase at subsequent intervals. The only exception is the measurement, but did not find a significant improvement of
score of the domain Speech Production (SP), where no pro- the sample’s total score after 1 year of CI use. In a figure
gress was made. While on the other domains, the scores included in their article, Louza et al. illustrate the pre- and
from intervals pre-operative and 24 months differed, on post-implantation NCIQ scores (total score and domain-
average, by some 10 points, on this domain, the two scores specific scores) of their sample. Indeed, when looking at
were identical. This finding suggests that speech produc- the total score, no pre–post change is seen. However, when
tion after cochlear implantation did not at all change in our looking at the domain-specific scores, a different picture
sample. This is not surprising, because SSD patients already emerges. One finds that in five out of six domains, the score
have good Speech Production before cochlear implantation. had increased after cochlear implantation by 5–10 points.
Hence, further improvement may not be expected. Only in one domain (Advanced Sound Perception) had it
When trying to compare our NCIQ findings to those of decreased by about 5 points. Considering these changes, one
other studies, one finds that comparability is limited. The wonders why they are not reflected in the total score. If the
major problem is that most studies that assessed HRQoL in scores of five out of six domains increase, and only one score
SSD patients did not use the NCIQ, but questionnaires such slightly decreases, then also the total score is expected to
as the APHAB, the GBI, the SSQ12, or the IO-HA. With increase. As we miss background information about Louza
regard to these studies, we can only state that our findings et al.’s study, we are not in the position to comment further
are in line with theirs in so far as most of them found that on their findings.
HRQoL (however measured) significantly improved in SSD
patients after cochlear implantation. Among the few stud- Generic health‑related HRQoL: HUI 3
ies that used the NCIQ, Häußler et al. [25] reported find-
ings that compare very well to ours. Häußler et al. admin- The HUI 3 utility scores of our sample showed a different
istered the NCIQ to a sample of 20 patients with a similar time course than the NCIQ scores. Both the single-attribute
degree of unilateral HL (up to 45 dB HL in the better ear) utility score (SAUS) of Hearing and the multi-attribute util-
and with a similarly wide range of duration of deafness ity score (MAUS) increased up to 6 months after implant
(0.3–27 years). Administration of the NCIQ occurred pre- activation, with the largest gains between 3 and 6 months
operatively and, on average, 1.5 years after implant surgery after implant activation. At subsequent time-points, how-
(range 0.5–3.3 years). The mean total score of their sample ever, both decreased again. The factor “Change over Time”
increased significantly from 63 to 74 points (+ 11 points) was significant for either scores: the MAUS and the SAUS.
after implantation. This increase corresponds well to that Post hoc comparisons between pre-operative and post-
observed in our sample (+ 9 points), even though in our operative scores did not show significant differences in all
sample, the total mean score was a little lower at the start- cases. This finding was somewhat unexpected in view of the
ing point and at the endpoint of the study. Also, Rösli et al. strong increase of the utility scores. It could be related to
[23] reported NCIQ scores from SSD patients. Their sample the hypothesis, that the Bonferroni adjustment (which was
consisted of eight SSD patients with normal hearing in the applied in our study) may be too conservative when a larger
better ear (PTA average: 7 dB HL) and an average duration number of subtests are performed. Nevertheless, the overall
of deafness of 7 ± 11 years. The NCIQ was administered, on increase of the scores between the time-points pre-opera-
average, 7–8 months after implant activation. The patients tive and 24 months after implant activation was impressive
indicated that, post-operatively, the scores had significantly (SAUS + 0.14; MAUS: + 0.09). Considering that an increase
increased (by > 10 points) on all domains of the NCIQ, of 0.05 is rated as clinically relevant, our findings suggest
except for Speech Production. Accordingly, the NCIQ total that cochlear implantation in SSD may be highly effective
score was significantly raised (+ 13 points). However, the in improving also the patients’ generic HRQoL.
large gains observed in this study must be seen with caution. It is remarkable that in the second half of the observa-
Because the study had a retrospective design, where pre- tion period the SAUS and the MAUS decreased. A possible
operative scores were not assessed but post-operatively esti- explanation could be that the questions of the HUI 3, which
mated, its results could be affected by a recall bias. Another assess the single attribute Hearing, are focusing on speech
study that used the NCIQ for assessment of HRQoL in SSD perception. The patients’ satisfaction with speech percep-
patients is the one of Sladen et al. [33]. This study also found tion, however, may vary over time. In the first months after
significant improvement on all six domains of the NCIQ implant activation, many patients are pleased when they
after 6 months of CI use. Unfortunately, NCIQ scores of notice the quick progress they are making. When asked for
this study are reported in a manner that does not allow direct the quality of their speech perception, they may indicate
comparison to our findings. a high level as they relate it to their previous experiences
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(without CI). After a year or so, however, the progress slows from the CI and that these benefits were comparable to those
down, and some patients may recognize that there are still experienced by patients with a short duration of deafness.
situations where they do not understand well. In reaction, The findings from our study support the view that
they may be frustrated, because their hopes were not ful- duration of deafness plays a complex role in SSD. On one
filled. When asked about the quality of their speech percep- hand, we observed that long-lasting deafness was a risk
tion, they now may indicate an only moderate level, as they factor for CI discontinuation. The two patients of our sam-
relate it to their previous hopes. This would explain why the ple, who discontinued CI use, had long-lasting deafness
utility scores of the HUI 3 declined a little after 1 year. In (> 10 years). Referring this number to the size of the sub-
contrast, NCIQ scores remained stable, because the ques- sample, it means that 18% of the patients with long-term
tions of the NCIQ cover a broader range of sound percep- deafness, but none of the patients with short-term deafness
tion, not only speech recognition in demanding situations. have become CI non-users.
On the other hand, the patients with long-term SSD who
continued using the CI experienced equal improvement of
Dropout rate HRQoL as the patients with short-term SSD. Either sub-
sample showed a significant post-implantation increase of
Our sample included 20 patients at study onset and 14 at HRQoL scores, with similar gains and a similar course
study end (24 months after implant activation). Hence, there over time. As for the generic HRQoL, it even appeared
was a loss of six patients corresponding to a 30% dropout that scores were higher in the subsample with long-lasting
rate. In four of the six patients (20%), the study dropout was deafness, but the interpretation of this finding is difficult.
not related to dissatisfaction with the CI (according to our At any rate, long-lasting deafness was not found to yield
information). Two patients, however, discontinued using the poorer HRQoL outcome after cochlear implantation in
CI during the study period (10% of the sample). The reasons SSD than short-lasting deafness.
for the discontinuation were that either they had problems Based on these observations, we conclude that the role
with adapting to the CI or that they did not experience the of long-term deafness for cochlear implantation in SSD
benefit expected from it. The latter point deserves special is indeed complex. Long-lasting deafness may require
attention as it could play a critical role for SSD patients’ stronger rehabilitative efforts to get the deaf ear reacti-
satisfaction with the CI. Because SSD patients have normal vated. Due to this, there is an enhanced risk that the patient
hearing in their intact ear, their expectations towards hear- becomes dissatisfied with the CI and discontinues using it.
ing with the implanted ear may be very high. Perhaps, they But in case the patient continues using the CI, long-term
hope to hear as well with it as with the intact ear. Finally, deafness is not a risk factor for poorer outcome of HRQoL.
they may be disappointed when they recognize that, despite In this regard, our findings add to those of other stud-
several months of auditory training, their hearing skills are ies that reported similar observations [37–39]. We hence
still below their expectations. Especially for patients with suggest that long-term SSD should not be considered a
long-lasting deafness, the reactivation of the deaf ear may contraindication against cochlear implantation. If patients
be a challenge [34]. Indeed, both patients of our sample who with long-term SSD adapt to the CI, they can draw sig-
discontinued CI use were unilaterally deaf since birth or nificant benefit from it and can obtain equal improvement
early childhood. Our findings are, therefore, supporting the as patients with short-term SSD. The important point is,
view that patients with long-lasting unilateral deafness are at however, that these patients are adapting to the CI—which
an enhanced risk of experiencing dissatisfaction with a CI. emphasizes the need for good rehabilitative support and
auditory training.
Outcome in patients with long‑lasting deafness
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SSD. In this regard, long-lasting SSD should not be con- longitudinal assessment of spatial hearing abilities and tinnitus
sidered a contra-indication for cochlear implantation. handicap. Otol Neurotol 35(9):1525–1532
12. Zeitler DM, Dorman MF, Natale SJ, Loiselle L, Yost WA,
Gifford RH (2015) Sound source localization and speech
Acknowledgements The authors thank Dr. Anke Hirschfelder, Charité understanding in complex listening environments by single-
University Clinic Berlin, for providing a German version of the NCIQ. sided deaf listeners after cochlear implantation. Otol Neurotol
36(9):1467–1471
Compliance with ethical standards 13. Mertens G, Kleine Punte A, De Bodt M, Van de Heyning P (2015)
Binaural auditory outcomes in patients with postlingual profound
Conflict of interests This study did not receive funding in financial unilateral hearing loss: 3 years after cochlear implantation. Audiol
form. The MedEl Elektromedizinische Geräte GmbH supported the Neurootol 20(Suppl 1):67–72
study with licencing of the HUI 3 utility tool. 14. Blasco MA, Redleaf MI (2014) Cochlear implantation in uni-
lateral sudden deafness improves tinnitus and speech compre-
Ethical approval This study was conducted according to the ethical hension: meta-analysis and systematic review. Otol Neurotol
standards of the 1964 Helsinki Declaration and its later amendments. 35(8):1426–1432
The study was approved by the Ethic Committees of the Medical Uni- 15. Tokita J, Dunn C, Hansen MR (2014) Cochlear implantation and
versity of Innsbruck, Austria, and the University of Würzburg, Ger- single sided deafness. Curr Opin Otolaryngol Head Neck Surg
many. 22(5):353–358
16. Vlastarakos PV, Nazos K, Tavoulari E-F, Nikolopoulos TP
Informed consent Informed consent was not applicable in this study (2014) Cochlear implantation for single-sided deafness: the out-
because data were obtained during routine clinical procedures. comes. An evidence-based approach. Eur Arch Otorhinolaryngol
271:2119–2126
17. Holder JT, O’Connell B, Hedley-Williams A, Wanna G (2017)
Cochlear implantation for single-sided deafness and tinnitus sup-
pression. Am J Otolaryngol 38(2):226–229
18. Mertens G, De Bodt M, Van de Heyning P (2016) Cochlear
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