Otology & Neurotology 36:1023Y1028 Ó 2015, Otology & Neurotology, Inc.

Real-World Verbal Communication Performance of Children Provided With Cochlear Implants or Hearing Aids *Hartmut Meister, †Annerose Keilmann, †Katharina Leonhard, ‡Barbara Streicher, *§Linda Mu¨ller, and ‡Ruth Lang-Roth *Jean Uhrmacher Institute for Clinical ENT-Research, University of Cologne, Cologne; ÞDepartment for Communication Disorders, University Medical Centre, University of Mainz, Mainz; þClinic of Otorhinolaryngology, Head and Neck Surgery, University of Cologne, Cologne; and §Department of Audiology and Phoniatrics, Charite´VUniversita¨tsmedizin Berlin, Berlin, Germany

Objective: To compare the real-world verbal communication performance of children provided with cochlear implants (CIs) with their peers with hearing aids (HAs). Study Design: Cross-sectional study in university tertiary referral centers and at hearing aid dispensers. Methods: Verbal communication performance was assessed by the Functioning after Pediatric Cochlear Implantation (FAPCI) instrument. The FAPCI was administered to 38 parents of children using CIs and 62 parents of children with HAs. According to the WHO classification, children with HAs were categorized into three groups (mildYmoderateYsevere hearing loss). Analysis of variance (ANOVA) was performed on the FAPCI scores, with study group, hearing age (i.e., device experience), and age at hearing intervention as sources of variation. Results: ANOVA showed that hearing age and study group significantly contribute to the FAPCI outcome. In all study

groups except the children with mild hearing loss, FAPCI scores increased alongside growing experience with the devices. Children with mild hearing loss using HAs showed higher scores than those with severe hearing loss or implanted children. There were no significant differences between the children with CIs and the children with moderate or severe hearing loss using HAs. Conclusion: Real-world verbal communication abilities of children with CIs are similar to those of children with moderate-tosevere hearing loss using amplification. Because hearing age significantly influences performance, children with moderate-tosevere hearing loss using HAs and implanted children catch up with children with mild hearing loss at a hearing age of approximately 3 years. Key Words: ChildrenVCochlear implantsV FAPCIVHearing aidsVVerbal communication performance. Otol Neurotol 36:1023Y1028, 2015.

Cochlear implantation has proved to be a beneficial, safe, and cost-effective measure to restore hearing in children with profound-to-severe hearing loss (1Y3). Many children provided with cochlear implants achieve openset speech perception after some time of implant use. Both speech perception and speech production abilities are prerequisites for the adequate verbal communication of the children, and research has shown that both domains develop with cochlear implant use, though sometimes

delayed in comparison to normal-hearing children and partly remaining behind the abilities of normal-hearing peers (4). The success of cochlear implantation in children depends on a number of factors, such as the psychosocial situation, engagement of the parents, educational environment, mode of communication, age at implantation, duration of deafness, or experience with the CI (5Y7). An intensively discussed question is whether early implantation yields a better outcome and if the potential benefits with regard to the development of communication abilities of the children outweigh the risks associated with very early implantation (8). Some studies have found an association between age at implantation and outcome with CI as reflected in a better outcome for speech perception skills and speech intelligibility (9,10) or more rapid development of speech production (11) for children implanted at an earlier age. Though there is no consensus

Address correspondence and reprint requests to Hartmut Meister, Ph.D., Jean Uhrmacher Institute for Clinical ENT-Research, University of Cologne, Geibelstr. 29Y31, D-50931 Cologne, Germany; E-mail: [email protected] The authors report no conflicts of interest. H.M. and A.K. equally shared authorship. A.K. received funding for investigator-initiated studies from Phonak and MED-EL. Supplemental digital content is available in the text.

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about the optimal age at intervention, it is suggested that implantation should take place before the second birthday (12,13). In addition to age at implantation, experience with the CI is a significant factor. This is typically called ‘‘hearing age’’ and covers the time span from hearing intervention to the time at which hearing or communication abilities are assessed. Age at fitting of the HA or CI and hearing age summarize to the chronological age of the child. Several studies have described developmental aspects of communication abilities in children using CIs, some of them using chronological age and others using hearing age as the baseline. May-Mederake et al. (14) examined the auditory development of children implanted with CIs by means of the LittlEARS inventory. LittlEARS (15) assesses the reception, understanding, and verbal production of acoustic (linguistic) stimuli in children younger than 2 years. May-Mederake et al. found that children using CIs reached the maximum possible score at an average hearing age of 22 months, which corresponded to 38 months of chronological age. A control group of normal-hearing children reached the maximum score by 24 months. The authors assumed a similar development in children early implanted with CI and their NH peers. Schramm et al. (16) conducted a longitudinal study with children who were implanted early (i.e., under 16 months of age) and normal-hearing children and assessed speech and language development over a period of 36 months after fitting of the devices. By means of different parentanswered questionnaires, they demonstrated that three of their five children with CIs reached similar scores as normalhearing children at a chronological age of approximately 18 months and showed even larger progress than their NH peers when results were related to individual hearing age. Assessment of speech perception development with respect to different age at implantation was the focus of an investigation by Tajudeen et al. (17). They found significantly higher word scores for the children who were implanted very early (G12 mo) as compared to the children implanted in the second or third year of life. However, when scores were expressed as a function of hearing age rather than chronological age, there were no significant group differences. Thus, chronological age should be considered as a variable or should be matched when comparing different groups with respect to the effect of age at intervention or the rehabilitation measure (i.e., cochlear implants or hearing aids). As technology has improved and effective rehabilitation programs are at hand, indication criteria for cochlear implantation have changed over the last years, thereby increasing the amount of pediatric patients provided with CIs (18). There is some debate about the criteria for cochlear implantation, especially if the children are provided with hearing aids that allow at least a certain degree of speech understanding. Blamey et al. (4) administered a number of speech perception, speech production, and language tests on 47 children provided with CIs and 40 children using amplification. When controlling for chronological age, the authors found little difference between

the two groups with respect to any of the measures. However, a negative trend in speech perception with increasing hearing loss was found in the children using amplification, whereas no such relationship was found for the language and speech production measures. When compared with regards to their unaided pure tone thresholds, it was reported that an ‘‘average’’ CI user with a hearing loss of 106 dB HL performed similarly to an ‘‘average’’ hearing aid user with a hearing loss of 78 dB HL. Similarly, Eisenberg et al. (19) compared the communication abilities of pediatric CI patients with children with hearing aids using a number of speech recognition and language measures. They found no differences between the two groups with the exception of the Peabody Picture Vocabulary Test, in which the children using amplification outperformed the children with CIs. However, this finding might also be accounted for by the fact that the children with HAs were substantially older than the implanted children and had more experience with their devices (i.e., greater ‘‘hearing age’’). Fitzpatrick et al. (20) examined children and adolescents aged 6 to 18 years using CIs or HAs with respect to their speech and language abilities. They found no differences between the two groups for the various speech recognition tests administered, but showed that the participants using amplification performed better than the CI users with respect to receptive vocabulary, language, and phonological memory. These investigations give interesting insight into the speech and language abilities of children provided with CIs or hearing aids based on clinical measures. Beyond this, the aim of the present study was to compare the realworld verbal communication abilities of children provided with CIs or HAs while considering hearing age and age at intervention as additional factors. Verbal communication abilities were assessed using the Functioning after Pediatric Cochlear Implantation (FAPCI [21]). A secondary aim of the present study was to consider different grades of hearing loss in the children using hearing aids. We hypothesized that children provided with cochlear implants show similar real-world verbal communication abilities as children with severe hearing loss using hearing aids.

METHODS Participants Participants were recruited within the framework of the clinical settings at the audiology centers involved. We included children with unilateral cochlear implants and bilateral hearing aids. This represented the basic clinical procedures at the time. Hearing aid fitting typically was based on the DSL method (desired sensation level [22]) that aims at placing amplified speech within the residual auditory area by considering individual characteristics of the child, such as real ear transfer functions. Children with multiple disabilities were excluded. Because the main aim of the study was to compare the communication performance of children with CIs or HAs, participants were selected from a larger population (n = 145) with respect to similar chronological age

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COMMUNICATION OF CHILDREN WITH COCHLEAR IMPLANTS (2 to maximum 6 yr) in all study groups. In total, the parents of 38 children with CI and 62 children with hearing aids were enrolled in the study. Forty-five children were female and 55 were male. The children with hearing aids were classified into three groups according to their degree of hearing loss. We used the classification of the WHO that applies somewhat coarser categories than, e.g., the ASHA classification. Following the WHO, the unaided better ear hearing loss (BEHL) averaged across the pure tone frequencies 0.5, 1, 2, and 4 kHz (four-frequency PTA) was used, dividing in groups of children with mild (BEHLG41 dB HL), moderate (BEHL between 41 and 60 dB HL), and severe (BEHL 960 dB HL) hearing loss. A group of children with profound hearing loss was not established because only two cases met the criterion BEHL980 dB HL. Thus, four study groups, namely children with hearing aids and mild (n = 15), moderate (n = 33), or severe hearing loss (n = 14) as well as children with cochlear implants (n = 38), were formed. The children were also characterized with respect to their age at intervention and their hearing age. Age at intervention was defined as the child’s age when the cochlear implant was activated or the hearing aid was fitted for the first time. Two groups were formedVchildren with intervention by the first 2 years of life (n = 59) or later (n = 41). With regard to hearing age, five groups were formed with hearing age less than 12 months (n = 17), between 12 and 23 months (n = 24), between 24 and 35 months (n = 31), between 36 and 47 months (n = 18), and greater than 47 months (n = 10). This classification was chosen because speech and language abilities of children with hearing loss show the greatest changes within the first 4 years of device usage (17). In general, the four study groups did not differ significantly with respect to age at intervention, hearing age, or chronological age (Kruskal-Wallistest, all p 9 0.05). Mean values and standard deviations are given in Table 1.

Questionnaire To assess the verbal communication performance of the children in everyday life, the parent-proxy Functioning after Pediatric Cochlear Implantation (FAPCI) questionnaire (21) was administered. The FAPCI was developed to complement tests of speech and language perception/production in children using CIs aged between about 2 and 5 years. Other inventories available in the German language, such as the LittlEARS questionnaire (15), address children of younger age (up to approximately 2 yr) or are not designed for the assessment of pediatric cochlear implantation. The FAPCI was originally developed for the assessment of children provided with CIs. However, as it presents items tapping into basic aspects of the verbal communication of children and normative data are available, the FAPCI is also suited for the assessment of children using amplification. The validated German version of the FAPCI (23,24) was used. It presents 23 items worded as statements (see Supplemental Digital Content, http://links.lww.com/MAO/A295). Using a five-point Likert scale, the respondents’ level of agreement was assessed for the statements and coded with 1 (no agreement) to 5 (complete

TABLE 1.

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agreement). Detailed information is available in Lin et al. (21) as well as in Grugel et al. (23,24).

Procedure Data were collected at two university audiology centers and two hearing aid dispensers involved in pediatric hearing rehabilitation. Parents who agreed to participate in the study received the questionnaire together with a prepaid self-addressed envelope. Parents covered a large range of different educational and socioeconomic backgrounds, and all had sufficient language skills to understand the content presented by the items of the questionnaire. All participants gave their written informed consent. The study protocol was approved by the local ethic committees.

Statistical Analysis FAPCI scores were subjected to an ANOVA (univariate general linear model), with study group, age at intervention, and hearing age as sources of variation. Bonferroni posthoc tests were used to confirm the significant effects found with the ANOVA. Pearson’s correlation coefficients were used to determine the relationship between hearing age and FAPCI scores. A normal distribution of the data was confirmed with the Kolmogorov-Smirnovtest. All analyses were performed with IBM SPSS Statistics 22. The level of significance was always set to p = 0.05.

RESULTS Data of the overall FAPCI score (sum of all items) for the children using CIs and for the three groups of children provided with amplification are shown in Figure 1. Scores are presented in relation to the individual hearing age of the child. In all study groups except the children with mild hearing loss, the FAPCI score increased significantly alongside hearing age, as demonstrated by Pearson’s correlation coefficients (CI: r = 0.49, p = 0.002; HA moderate HL: r = 0.36, p = 0.042; HA severe HL: r = 0.84, p G 0.001). The children with mild hearing loss did not show such a relationship, as most scores were in the upper range of the FAPCI (maximum possible score 115 points) regardless of hearing age. Subjecting the FAPCI scores to an ANOVA with study group, age at intervention, and hearing age as sources of variation revealed a significant influence of hearing age (F4,100 = 4.32, p = 0.004) and study group (F3,100 = 2.72, p = 0.05). No other main effects or interactions were significant. Posthoc tests (Bonferroni) confirmed the significant effect of hearing age: the group of children with a hearing age greater than 47 months showed significantly higher scores than the group of children with a hearing age less than 12 months (p = 0.039) and those with a hearing age between 12 and 23 months (p = 0.006).

Mean and standard deviation (parentheses) of age at intervention, hearing age, and chronological age of the four study groups

Age Age at intervention, mo Hearing age, mo Chronological age, mo

CI n = 38

HA Mild HL n = 15

HA Moderate HL n = 33

HA Severe HL n = 14

23 (12) 26 (14) 50 (15)

26 (21) 28 (14) 55 (11)

23 (18) 25 (16) 48 (13)

14 (10) 35 (15) 50 (18)

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FIG. 1. Overall FAPCI scores as a function of hearing age for the children provided with cochlear implants (CI, n = 38), children with severe hearing loss (n = 14), children with moderate hearing loss (n = 33), and children with mild hearing loss (n = 15) using hearing aids (HA). Circles depict age at intervention (AI) below 24 months, triangles AI Q 24 months. Dashed lines indicate lower and upper boundaries for the FAPCI scores; solid lines indicate linear regression functions.

Children with hearing age between 36 and 47 months showed significantly higher scores than the group of children with hearing age less than 12 months (p = 0.037) and those with hearing age between 12and 23 months (p = 0.003). Posthoc tests (Bonferroni) also confirmed the significant effect of study group. The children with mild hearing loss showed significantly higher scores than the children with severe hearing loss (p = 0.008) and those provided with CI (p = 0.003). Mean FAPCI scores were 77 T 26 (CI), 74 T 24 (HA severe HL), 85 T 25 (HA moderate HL), and 101 T 9 (HA mild HL). Age at intervention did not show a significant main effect or any significant interaction.

DISCUSSION This study extends previous research on pediatric cochlear implantation by comparing the real-world verbal communication abilities of children provided with CI with children using amplification. All children were between 2 and 6 years of age, and the groups were matched with respect to chronological age to avoid bias resulting from gross age differences. Data analysis considered age at intervention and hearing age (i.e., device experience) of the children. Hearing age had a significant main effect on the overall scores of the FAPCI. Children with more experience showed better results than children with a low hearing age. This was

evident for three of the four study groups: a significant positive correlation could be found for all children except those with mild hearing loss, who mostly scored in the upper range of the FAPCI regardless of hearing age. Recalling that these children had averaged pure tone hearing losses below 41 dB, the concept of hearing age might be misleading in this group. It can be argued that these children received a certainValthough not adequateVauditory input before hearing aid fitting, which might explain their good verbal communication abilities even after a short duration of HA usage. In children with CI or hearing aids and moderate hearing loss, a significant correlation was evident between the FAPCI scores and hearing age, although the variance was rather largeVespecially for the children with a low hearing age. When looking at the data in the range of hearing age up to approximately 24 months, a number of children in both groups perform near the ceiling, whereas others show low scores. That is, individual children start on a rather low level but might show good communication abilities if they have more experience. Though this study is based on cross-sectional rather than on longitudinal data, this might provide some evidence that the children in these two study groups catch up with those with mild hearing loss with a higher hearing age of approximately 3 years (Fig. 1). The strongest relationship between the FAPCI scores and hearing age could be observed in the children with severe hearing loss using amplification. Hearing age explained a large proportion of variance in the scores

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COMMUNICATION OF CHILDREN WITH COCHLEAR IMPLANTS (about 70%), and the observation of large scatter with low hearing ageVas evident in the other two study groupsV could not be made because all children with a low hearing age had low or moderate scores. These findings might be due to the fact that some of the children with CI with high FAPCI scores and low hearing age had benefited from amplification before CI provision. Other factors might be different family support level or educational mode. However, these aspects were not explicitly assessed with the present study. The large variability of the FAPCI scores with low hearing age in the children with moderate hearing loss using amplification might to a certain extent be attributed to their individual hearing loss because a significant correlation (r = j0.51, p = 0.036) could be found between the four-frequency PTA and the FAPCI score when children with hearing age 24 months or less were considered. Analysis of the FAPCI scores also revealed a significant (p = 0.05) influence of the study group. Posthoc tests confirmed a significantly higher mean score for the children with mild hearing loss using amplification than for the children with severe hearing loss or CI. However, following the results presented above, it seems that the group difference occurred mainly because the children with mild hearing loss revealed high scores even if they had a low hearing age. It was also evident that the children of the other groups catch up with those children when they gain more experience with their devices. In the present investigation, no significant differences in the outcome of children provided with CI and the HA users with moderate-to-severe hearing loss (WHO classification) could be found. We are not aware of any study that compared the communication abilities of HA users with different grades of hearing loss with children provided with CIs. Other studies (4,19,20) suggest that pediatric CI users and children with severe-to-profound hearing loss using amplification show similar results in several speech and language domains. The children in these studies had mean three-frequency (500 Hz, 1 kHz, 2 kHz) PTAs of 78 dB HL (4), 78 dB HL (19), and 69 dB HL (20). The authors argued that this confirms current indication criteria of cochlear implantation in children, as the outcome of the groups of HA and CI users were similar with regards to different speech and language measures. The children using amplification enrolled in the present study had mean four-frequency PTAs of 28 dB HL (mild), 50 dB HL (moderate), and 74 dB HL (severe). Corresponding three-frequency PTAs were 27, 48, and 71 dB HL. FAPCI scores of the children with CI and those with severe hearing loss were almost identical (77 to 74 points). The children with moderate hearing loss also did not show a score significantly different from that of the children with CI. Following this line of argumentation, one could claim that children with CI perform similar to children using amplification with an average hearing loss of about 50 dB HL. However, it must be considered that the children with moderate hearing loss showed a clearlyV albeit not significantlyVhigher FAPCI score (85 points) and that comparison was based on only 71 data sets.

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Age at intervention did not show a significant influence on the outcome. This seems to stand in contrast to other studies that report a greater benefit for children with early implantation over children with later interventions (7,25,26). This might be due to different reasons: first, when having a closer look at studies that reported better outcomes with earlier intervention, these often describe developmental aspects with the trajectory of aural rehabilitation. Niparko et al. (27) reported steeper rate increases in both speech comprehension and speech expression for a younger age upon cochlear implantation. Grugel et al. (24) showed that children with early implantation followed the development of normal-hearing peers more closely than children with later implantation. In line with this, Dunn et al. (28) showed that the effect of age at implantation diminished with time, which held particularly true for higher-order skills such as language and reading. Thus, when individual communication development remains unconsideredVas is the case with the cross-sectional data in the present studyVthe beneficial effect of early implantation might not be fully accounted for. Second, Harrison et al. (10) have shown that the age at intervention, which best separates performance of children with early implantation versus late implantation, strongly depends on the test applied. For example, the Test of Auditory Comprehension (TAC) revealed an optimum split at an age at intervention of 4.4 years, whereas the optimum split of the more complex Phonetically Balanced Kindergarten (PBK) word list was at 8.4 years. The psychometric properties of the FAPCI with regards to the optimal separation of early versus latertreated children are unknown. Third, age at intervention is typically defined as the time at which the child’s hearing is essentially ‘‘turned on’’ (10). This concept is valid for cochlear implantation but might be misleading when the child has received a substantial amount of auditory stimulation before intervention, as evident at least with mild hearing loss. However, reanalyzing the data with excluding the study groups of children with mild and/or moderate hearing loss also showed no significant effect. Finally, sample size of the present study may only allow for assessment of larger effects associated with different age of intervention.

CONCLUSION Real-world verbal communication abilities of children with cochlear implants and hearing aids are significantly related to the hearing age of the children. This reflects developmental aspects of verbal communication connected to experience with the hearing device. Children with mild hearing loss using amplification outperform children with severe hearing loss using HAs and those using CIs. However, these differences disappear with a higher hearing age of approximately 3 years. Children with CI show very similar FAPCI scores compared to children with moderateto-severe hearing loss using hearing aids. Otology & Neurotology, Vol. 36, No. 6, 2015

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Real-World Verbal Communication Performance of Children Provided With Cochlear Implants or Hearing Aids.

To compare the real-world verbal communication performance of children provided with cochlear implants (CIs) with their peers with hearing aids (HAs)...
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