Abstract
Cochlear implantation (CI) has been successful in individuals with sensorineural hearing loss (SNHL) over the last few years. This study assessed the auditory and speech performance results of pediatrics with inner ear malformations (IEMs) following cochlear implantation at the Ahvaz cochlear implantation center and compared the outcomes of various malformations. All pediatrics with IEMs undergoing CI were included in the study. This retrospective study was performed on pediatrics with congenital IEMs who underwent cochlear implantation in Ahvaz cochlear implantation center between 2014 and 2019. The Category of Auditory Performance (CAP) and the Speech Intelligibility Rating (SIR) scores are two of the most frequently administered tests. The CAP with ranging from 0 (displays no awareness of environmental sounds) to 7 (can use the telephone with a familiar talker), was used to measure the speech perception performance of the implanted children. Furthermore, SIR consists of five performance categories ranging from “prerecognizable words in spoken language” to “connected speech is intelligible to all listeners”. Finally, the study included 22 patients. The evaluation of the CT-Scan revealed three types of inner ear malformation: Incomplete Partition (IP)-I in 2 (9.1%), IP-II in 12 (54.5%), and common cavity in 8 (36.4%) individuals. The results were shown that the median CAP score preoperative was 0.5 (interquartile range (IQR): 0–2) and postoperative was 3.5 (IQR: 3–7). There were statistically significant differences in CAP scores between preoperative and second-year follow-up postoperative (p value = 0.036). The results were shown that the median SIR score preoperative was 1 (IQR: 1–5) and postoperative was 2 (IQR: 1–5). There were statistically significant differences in SIR scores between preoperative and second-year follow-up postoperative (p value = 0.001). Following a thorough preoperative screening, patients with specific IEMs can be candidates for CI and not a contraindication. There were statistically significant differences in CAP and SIR scores between preoperative and second-year follow-up postoperative in the common cavity and IP-II groups.
Keywords: Sensorineural hearing loss, Cochlear implant, Inner ear malformation, Pediatrics
Introduction
In recent years, cochlear implantation (CI) has proven effective for pediatric patients with sensorineural hearing loss (SNHL) [1]. For pediatric patients with severe hearing loss, CIs have evolved into a life-altering technology [2]. Currently, there are multiple preoperative methods for assessing the cochlear structures following CI. Unfortunately, no global consensus on recommended tests makes comparisons across various populations difficult [3, 4]. The Category of Auditory Performance (CAP) and the Speech Intelligibility Rating (SIR) scores are two of the most frequently administered tests [4, 5], in addition, computed tomography (CT) and magnetic resonance imaging (MRI) can be used to evaluate cochlear structures [6].
Pediatric patients with inner ear malformations (IEM), which account for roughly 20–35% of cases of congenital SNHL [2, 7], are not typically advised to use CIs. Unexpected effects following CI cannot be predicted in cases of IEM because the neural tissue distribution and atypical positions of anatomical markers such as the lateral semicircular canal and facial nerve may make surgery difficult [8]. Complete Labyrinthine Aplasia or Michel Deformity is characterized by the absence of the cochlea, vestibule, semicircular canals (SCCs), and vestibular and cochlear aqueducts. Rudimentary Otocyst (millimetric representations of the otic capsule in the absence of an internal auditory canal (IAC)), also referred to as a rudimentary otocyst. Enlarged Vestibular Aqueduct, Cochlear aplasia (the lack of a cochlea), Common Cavity (a single, ovoid or circular chamber representing the cochlea and vestibule), Incomplete Partition of the Cochlea I-II (IP-I and IP-II), Cochlear Hypoplasia, and Cochlear Aperture Abnormalities are the eight categories in the IEM [9, 10]. Numerous pediatric patients with IEM are candidates for a CI or an auditory brainstem implant (ABI) due to their bilateral severe to profound hearing loss [11]. Specific research indicates that the efficacy of CIs is diminished during severe IEM [12, 13]. Nevertheless, numerous studies have demonstrated that CI benefits these patients; the results vary depending on the abnormality [14–16].
Despite the complications and potential surgical challenges, the outcomes of CI surgery are comparable to those of patients with congenital malformation. The existence of IEM raises several issues concerning the optimal surgical method and electrode, the identification of prognostic variables, the impact of comorbidities, and the risk of complications [12, 17]. However, since IEM occurs in up to 20% of children with SNHL and some surgeons may be hesitant due to potential surgical complications, it is essential to provide an update on the results [8, 18].
This study compared the outcomes of various malformations to the auditory and speech performance results of pediatrics with IEM following cochlear implantation at the Ahvaz cochlear implantation center.
Materials and Methods
Study Design and Setting
This retrospective study was performed on pediatrics with congenital IEMs who underwent cochlear implantation in Ahvaz cochlear implantation center between 2014 and 2019. Ethics approval was granted by the Research Ethics Committee of Ahvaz Jundishapur University of Medical Sciences (IR.AJUMS.HGOLESTAN.REC.1399.130). This investigation was also conducted following Helsinki's ethical approval for human studies. We obtained written agreement from all participants or their parents, and if any individuals were unwilling to continue the study, they were omitted from the analysis.
Participants
All pediatrics with IEMs undergoing right-side CI were included in the study. Also, patients with cochlear nerve agenesis, every conductive disorder, cochlear agenesis, Inability to follow up after the operation, and no consent to inform were excluded from this study.
Variables
By evaluating the HRCT scans, the IEM was identified. The more severe abnormality was addressed first, and the consequence of this concurrency was assessed separately. The CAP and SIR scores were obtained before operation and the second year afterward by retrospectively reviewing the patients' files. The CAP with ranging from 0 (displays no awareness of environmental sounds) to 7 (can use the telephone with a familiar talker), was used to measure the speech perception performance of the implanted children. Furthermore, SIR consists of five performance categories ranging from “prerecognizable words in spoken language” to “connected speech is intelligible to all listeners”. The difference between the second-year follow-up and the preoperative scores was used to calculate the auditory and speech performance.
Data Sources/Measurement
Auditory examinations were done to determine the presence and degree of hearing loss. Clinical examinations such as auditory brainstem response (ABR), acoustic reflex and emission, age-appropriate behavioral audiometry, and tympanometry were used to assess the existence and severity of hearing loss. An otorhinolaryngologist and a detailed pediatrician history and physical examination to examine the concomitant malformations. Otologists and radiologists identified IEMs separately using a high-resolution computed tomography (HRCT) scan of the temporal bone [19].
Before the operation, all patients had auditory therapy for at least three months, with little or no improvement. Trans-orbital X-ray was used to check the electrode's location after the operation. Four to six weeks following the process, the CIs were activated and installed.
Expert rehabilitation therapists employed the CAP index to evaluate the auditory skills of pediatrics preoperatively the second year after CI operation [3]. Expert speech therapists used the SIR scale to assess speech intelligibility in pediatrics preoperatively during the second year following operation [5].
Study Size
Due to the rarity of these anomalies, all cases of abnormalities were selected and studied.
Statistical Methods
The SPSS version 25 software (Chicago, USA) was used for data analysis. The descriptive statistical methods were used, including frequency tables, medians, interquartile ranges (IQR)s, and appropriate scattering such as mean and standard deviation (± SD). The quantitative normality data was checked by Kolmogorov–Smirnov test. To compare the quantities before and after, Kruskal-Wallis with Dunn test post hoc, comparison of quantities between several groups, one-way analysis of variance. The statistical significance level of the tests was less than 0.05%.
Results
Participants
Finally, the study included 22 patients (10 girls) were included. At the hearing loss diagnosis, the average age was 58.50 ± 35.2 months (20 to 152 months). The evaluation of the CT-Scan revealed three types of inner ear malformation: IP-I in 2 (9.1%), IP-II in 12 (54.5%), and common cavities in 8 (36.4%) individuals. All pediatrics completed the second-year follow-up assessment (Table 1, Fig. 1, and 2).
Table 1.
Distribution of patients in groups
| Variables | Common cavity (N = 8) |
Incomplete partition type 1 (IP−1) (N = 2) | Incomplete partition type 2 (Mondini) (IP-2) (N = 12) | p value | |
|---|---|---|---|---|---|
|
Age (Month), Mean ± SD (Min–Max) |
87.5 ± 41.18 (40–152) |
35 ± 8.48 (29–41) |
43.08 ± 17.57 (20–80) |
0.078 | |
| Gender, N (%) | Boy | 4 (50) | 2 (100) | 6 (50) | 0.9 |
| Girl | 4 (50) | – | 6 (50) | 0.15 | |
Fig. 1.
Incomplete partition type II or Mondini’s malformation. Axial high-resolution multidetector CT image of temporal bone shows an incomplete partition of the cochlea with normal basal turn and cystic apices with 1.5 turns. Dilatation of the vestibule and vestibular aquiduct is also seen
Fig. 2.
Common cavity malformation. Axial high-resolution multidetector CT image of temporal bone shows the confluence of the cochlea, vestibule, and semicircular canals formed a common cavity
The Auditory Performance
The results were shown that the median CAP score preoperative was 0.5 (IQR: 0–2) and postoperative was 3.5 (IQR: 3–7). There were statistically significant differences in CAP scores between preoperative and second-year follow-up postoperative (p value = 0.036) (Table 2). In patients with common cavity malformations, the median CAP score preoperative was 0, and second-year postoperative was 3.5, which has improved and is statistically significant (p value = 0.011). Also, in patients with IP-II malformations, the median CAP score preoperative was 1, and second-year postoperative was 4.5, which has improved and is statistically significant (p value = 0.003). Also, in patients with IP-I malformations, the median CAP score preoperative was 1.5, and the second-year postoperative score was 3.5, which has improved (Table 2).
Table 2.
Auditory performance scores (CAP) score before surgery and second year postoperatively
| Patient | CAP Pre op | CAP Second year Post op | P value |
|---|---|---|---|
| Common cavity, Median and IQR | 0 (0–1) | 3.5 (3–7) | 0.036* |
| Case 1 | 0 | 3 | |
| Case 2 | 1 | 3 | |
| Case 3 | 0 | 4 | |
| Case 4 | 0 | 7 | |
| Case 5 | 1 | 3 | |
| Case 6 | 0 | 5 | |
| Case 7 | 0 | 3 | |
| Case 8 | 0 | 4 | |
| Incomplete partition type 1 (IP-1), Median and IQR | 1.5 (1–2) | 3.5 (3–5) | |
| Case 1 | 2 | 5 | |
| Case 2 | 1 | 3 | |
| Incomplete partition type 2 (Mondini), Median and IQR | 1 (0–2) | 4 (0–7) | |
| Case 1 | 1 | 5 | |
| Case 2 | 2 | 7 | |
| Case 3 | 2 | 7 | |
| Case 4 | 2 | 5 | |
| Case 5 | 0 | 3 | |
| Case 6 | 1 | 3 | |
| Case 7 | 1 | 3 | |
| Case 8 | 1 | 5 | |
| Case 9 | 0 | 0 | |
| Case 10 | 0 | 4 | |
| Case 11 | 0 | 1 | |
| Case 12 | 0 | 5 | |
| P value | 0.081* | 0.883* |
*Kruskal-Wallis with Dunn test post hoc
The Speech Production
The results were shown that the median SIR score preoperative was 1 (IQR: 1–5) and postoperative was 2 (IQR: 1–5). There were statistically significant differences in SIR scores between preoperative and second-year follow-up postoperative (p < 0.001) (Table 3). In patients with common cavity malformations, the median SIR score preoperative was 1, and second-year postoperative was 2, which has improved and is statistically significant (p value = 0.034). Also, in patients with IP-II malformations, the median SIR score preoperative was 1, and second-year postoperative was 2, which has improved and is statistically significant (p value = 0.016). Also, in patients with IP-I malformations, the median SIR score preoperative was 1, and the second-year postoperative score was 1.5, which has improved (Table 3).
Table 3.
Speech intelligibility rating (SIR) score before surgery and second-year postoperatively
| Patient | SIR Pre op | SIR Second year Post op | P value |
|---|---|---|---|
| Common cavity, Median and IQR | 1 (1–5) | 2 (1–5) | p < 0.001 |
| Case 1 | 1 | 1 | |
| Case 2 | 1 | 2 | |
| Case 3 | 1 | 2 | |
| Case 4 | 5 | 5 | |
| Case 5 | 1 | 3 | |
| Case 6 | 1 | 2 | |
| Case 7 | 1 | 1 | |
| Case 8 | 1 | 2 | |
| Incomplete partition type 1 (IP-1), Median and IQR | 1 (1–1) | 1.5 (1–2) | |
| Case 1 | 1 | 2 | |
| Case 2 | 1 | 1 | |
| Incomplete partition type 2 (Mondini), Median and IQR | 1 (1–1) | 2 (1–4) | |
| Case 1 | 1 | 3 | |
| Case 2 | 1 | 3 | |
| Case 3 | 1 | 4 | |
| Case 4 | 1 | 2 | |
| Case 5 | 1 | 1 | |
| Case 6 | 1 | 2 | |
| Case 7 | 1 | 1 | |
| Case 8 | 1 | 2 | |
| Case 9 | 1 | 1 | |
| Case 10 | 1 | 1 | |
| Case 11 | 1 | 1 | |
| Case 12 | 1 | 2 | |
| p value | 0.417* | 0.674* |
*Kruskal-Wallis with Dunn test post hoc
Discussion
Despite the success of CI in pediatrics with IEM, the results varied. This study sought to evaluate the impact of CI on the auditory and speech production systems. Some studies have demonstrated that, except for cochlear nerve aplasia, CI is a safe and beneficial procedure [11, 20, 21].
After a two-year follow-up of postoperative IEM patients, this study revealed that patients with common cavity malformations who underwent CI, CAP, and SIR scores improved significantly. To this end, Daneshi et al. (2020) demonstrated that the median CAP and SIR ratings improved significantly within the first two years following CI in both groups [11]. This study showed no significant difference between the CAP and SIR scores of children with inner ear abnormalities. Our study revealed statistically significant differences in CAP and SIR scores between preoperative and two-year postoperative follow-up for all abnormality groups. Moreover, Pradhananga et al. (2015) evidenced that after more than three years of follow-up, the CAP score in patients with common cavity malformations increased to a mean of 5.0, whereas the SIR score only marginally changed [22].
Although the severity of the malformation treated with CIs appears to be the most significant factor influencing hearing perception in IEMs patients, several other variables such as the patient's age, the time of cochlear implant, and the hearing rehabilitation may also play a significant role in these results [8, 23]. Indeed, CI improves auditory perception and communication skills in pediatric patients with a common cavity, although the audiological improvement was not as significant as in those without a common cavity [24, 25]. This study also showed that CAP and SIR scores improved significantly in patients with Incomplete partition type II (Mondini) malformations who underwent cochlear implantation. Similarly, other studies demonstrated a statistically significant difference between postoperative and preoperative CAP and SIR scores in patients with IP-II. In the study by Daneshi et al. (2020), patients exhibited increases of 4 points in CAP and 3 points in SIR for IP-II [11]. However, this rating score in our study was 3 points lower in CAP, and 1 point lower in SIR following a two-year follow-up, and these differences may be attributable to the duration of the follow-up. In a systematic review, Farhood et al. (2017) observed that patients with IP-II demonstrated the best outcomes. On the other hand, patients with common cavities and cochlear hypoplasia exhibited less improvement [14]. In addition, another study found variable statistics ranging from good to poor performance CI for IP-II patients [26].
Due to the small number of patients with IP-I malformations, however, accurate conclusions cannot be drawn from the results. At the same time, the CAP and SIR scores improved during the second year of follow-up. Accordingly, Eftekharian et al. (2019) studied 18 patients with IP-I anomalies who underwent CI. They observed that cochlear implantation in these patients was safe and effective [27].
Overall, CI is safe for patients with IEM, but its success rate is lower than that of patients with normal inner ear anatomy [28] ; however, it can still be considered to improve the quality of life of these patients [22].
Limitations
This study was run in one cochlear implant center without a control group for comparison.
Conclusion
Following a comprehensive preoperative screening, patients with specific IEM may be candidates for CI and are not contraindications. In the common cavity and IP-II groups, there were statistically significant differences in the CAP and SIR scores between preoperative and two-year postoperative follow-up.
Acknowledgments
The Office of Vice-chancellor financially supported this study for Technology and Research Development of Jundishapur Medical Sciences University, Ahvaz, Southwest Iran, as part of Mostafa Timar’s thesis under the research code HRC-9914.
Author Contributions
All authors (MT, NS, AB, MH) made substantial contributions to the study’s conception, design, data acquisition, analysis, and/or interpretation; participated in drafting the manuscript or revising it critically for substantial intellectual content; agreed to submit the manuscript to this journal; gave final approvals of the submitted version; and agreed to be accountable for all aspects of this publication.
Funding
Ahvaz Jundishapur University of Medical Sciences number: HRC-9914.
Data Availability
The datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request.
Declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical Approval
This study was reviewed and approved by the Ahvaz Jundishapur ethical comities (Ethic code: IR.AJUMS.HGOLESTAN.REC.1399.130). The study was conducted in compliance with the principles of the Declaration of Helsinki and Good Clinical Practice guidelines. All subjects provided written informed consent to participate in the study. All authors (Mostafa Timar, Nader Saki, Arash Bayat, Mohammadghasem Hanafi) made substantial contributions to the study’s conception, design, data acquisition, analysis, and/or interpretation; participated in drafting the manuscript or revising it critically for substantial intellectual content; agreed to submit the manuscript to this journal; gave final approvals of the submitted version; and agreed to be accountable for all aspects of this publication.
Footnotes
Publisher’s Note
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Data Availability Statement
The datasets generated during and/or analyzed during the current study are available from the corresponding author on reasonable request.