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PEDOT-7511; No. of Pages 4 International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Outcomes after cochlear reimplantation in children Franc¸oise Sterkers, Fanny Merklen, Jean Pierre Piron, Adrienne Vieu, Fre´de´ric Venail, Alain Uziel, Michel Mondain * Cochlear Implant Center Montpellier-Palavas, University Hospital of Montpellier, 34295 Montpellier Cedex 5, France
A R T I C L E I N F O
A B S T R A C T
Article history: Received 26 November 2014 Received in revised form 16 February 2015 Accepted 17 March 2015 Available online xxx
Objectives: With cochlear implantation now a routine procedure, reimplantation is becoming more commonplace for medical/surgical complications or device malfunctions. This study investigated the indications for reimplantation and the auditory outcomes following reimplantation surgery in prelingually-deafened children. Methods: Of the 539 prelingually deafened children implanted between 1990 and 2013, 45 were reimplanted (8.3% of implantations). Causes of reimplantation, type of device and angle of insertion at initial implantation were recorded, as well as type of implant reinserted, number of electrodes inserted and angle of insertion (calculated on cone beam computed tomography) on reimplantation, and finally any surgical findings. Speech perception test scores (phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores) were obtained 1, 2 and 3 years after reimplantation, and compared against the best speech recognition score obtained with the first implant before failure. Results: Medical reasons for reimplantation were found in 10 cases (22.2%). A malfunctioning device had occurred in 35 cases (77.7%) including hard failure in 24 and soft failure in 11. Complete insertion was achieved in the scala tympani in 42 cases and in the scala vestibuli in one case; partial insertion occurred in the remaining two cases. In two cases, one or two electrode rings snatched off from the electrode array during removal. The mean insertion angle was 330.58 before surgery and 311.88 after reimplantation (no statistical difference p = 0.48). The postoperative speech perception outcome measures showed no significant difference to the best score before reimplantation. Angle of insertion, type of device and etiology of deafness did not influence the results. The PBK performance improved over 10% in 43.2% of children, was similar in 40.5%, and showed a more than 10% decrease in 16.2% of children after reimplantation. The latter decline in performance was explained for some children by a partial insertion. Conclusions: Reimplantation has no negative effect on auditory outcome. In rare cases, speech perception outcome may not improve, requiring a specific rehabilitation program. ß 2015 Elsevier Ireland Ltd. All rights reserved.
Keywords: Cochlear implant Child Reimplantation Device failure
Cochlear implantation has become a routine procedure for restoring serviceable hearing to children having suffered a bilateral severe-to-profound sensorineural hearing loss with little or no benefit from hearing aids. In some cases, a reimplantation surgery is mandatory. The reasons for cochlear implant explantation or reimplantation can be divided into medical or surgical complications and device malfunctions. Device malfunctions comprise hard
* Corresponding author. Tel.: +33 467336804. E-mail addresses: [email protected], [email protected] (M. Mondain).
and soft failures and account for the majority of reimplantation surgeries. Several studies have investigated the indications, complications and results of reimplantation [1–10]. Most of these included both adults and children and showed no detrimental effect on speech perception outcome of reimplantation. Few specific pediatric series has been published recently [11,12], yet children may have a slightly higher rate of reimplantation, believed due to a higher incidence of trauma among children than in adults. This study aimed to investigate the indications for reimplantation, to assess the auditory outcomes following reimplantation surgery in prelingually deafened children, and to identify factors that may influence these outcomes.
http://dx.doi.org/10.1016/j.ijporl.2015.03.015 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
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PEDOT-7511; No. of Pages 4 F. Sterkers et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
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1. Materials and methods Medical reports from a total of 539 prelingually deafened children implanted between 1990 and 2013 were retrospectively analyzed. Overall, 45 cochlear implants had been replaced (8.3% of the implantations). Age at initial implantation ranged from 1.1 to 14.9 years (mean 5.07 years). Twenty boys and 25 girls aged from 5.2 to 22.8 years (average 13.9 years) underwent a reimplantation. Time from initial implantation to reimplantation ranged from 5 months to 21.7 years. Etiologies, detailed in Table 1, were primarily genetics-related (29%), meningitis (13%), and unknown (31%). Medical records were reviewed to identify causes of reimplantation, type of device and length of insertion at initial implantation, type of implant reinserted, number of electrodes inserted and angle of insertion (calculated on cone beam computed tomography [13]) on reimplantation, and surgical findings. The primary outcome measures were speech discrimination measured using phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores. Speech perception test scores were obtained 1, 2 and 3 years after reimplantation, and compared to the best results obtained with the first implant before failure. For individual patients, an increase in PBK scores between the first and second implantation of 10% or greater was regarded as an improved performance. Similarly, a reduction in PKB scores of 10% or more was regarded as a deterioration in performance. Statview software (Abacus1) for PC was used for statistical analysis. Statistical differences were analyzed using a non parametric test for paired data (Wilcoxon’s rank test). Statistical significance was set at the 5% level. Correlations were analyzed using ANOVA analysis.
2. Results 2.1. Type of device Three manufacturers were used for implantation: Cochlear, Neurelec and Advanced Bionics. Table 2 indicates the specific distribution of cochlear implant models (Cochlear 80% of population, Neurelec 4.5%, AB 15.5%) that required reimplantation and that of the models used to replace them. Forty-four reimplantations were performed with a device from the same company. The vast majority of Nucleus CI 22 were upgraded with a Nucleus CI 24 device, while the vast majority of the Clarion 1.2 CI were upgraded with a Clarion HF 90 CI. Table 1 Etiology of deafness for the 45 reimplanted children. N
Etiology
%
Genetics—nonsyndromic
GJB2 or GJB6 gene mutation Other
4 9
9 20
Genetics—syndromic
Usher type 1 Waardenburg Down syndrome Landau–Kleffner Mohr–Tranebjaerg syndrome
5 1 1 1 1
11 2 2 2 2
Meningitis Labyrinthitis CMV
6 1 1
13 2 2
1
2
14
31
Infectious
Perinatal Anoxia Unknown
Table 2 Distribution of the models of cochlear implants at implantation and at reimplantation. CI model
At implantation
At reimplantation
Nucleus CI22 Nucleus CI24 (M–R–RE) Nucleus CI512 Clarion 1.2 Clarion HF 90 Digisonic
24 10 2 6 1 2
3 33 1 0 6 2
2.2. Reasons for reimplantation Medical reasons accounted for the reimplantation in 10 cases (22.2%). These were infection in four cases all related to flap problems, head trauma with decreasing performance in another four, and neurologic problems in two. One of these latter children had Landau–Kleffner syndrome without improvement of language skills despite a 20% correct score on open-set recognition tests, and the other child required deep brain stimulation to control a severe dystonia (Mohr–Tranebjaerg syndrome). Malfunctioning device accounted for the decrement in clinical performance found in 35 cases (77.7%). This was a hard failure (abnormal integrity testing) in 24 cases (53.3%) and a soft failure (normal integrity testing though poor performance from the implant and/or nonauditory symptoms) in the remaining 11 (24.4%). 2.3. Surgical findings All reimplantations were performed at the same side, with one patient undergoing bilateral implantation during the reimplantation surgery. We achieved complete insertion in the scala tympani in 42 cases (93.3%). Partial insertion occurred in two cases: one child with unknown etiology of deafness with partial insertion at reimplantation (Nucleus CI24M; 12 active electrodes outside the cochleostomy), and the other child deafened from meningitis in whom insertion was partial during both implantation and reimplantation (Nucleus CI 512; 6 active electrodes outside the cochleostomy at reimplantation–ossification of the basal turn with partial insertion at implantation). Reimplantation was easy in all other cases of post-meningitis deafness, even in a child with round window ossification at implantation. In one case, we performed a complete insertion in the scala vestibuli at reimplantation. In two cases, electrode rings were snatched off the electrode array during removal and were left in place (one apical electrode ring snatched off a Digisonic SP device in one child, and two apical electrodes in another implanted with a Nucleus CI22 device): complete insertion of the new electrode array was achieved in both cases. Three cases required a complementary tympanoplasty (one eardrum perforation–two tympanic membrane retractions). No reimplantation was performed in cochlear abnormality cases (two children has an enlarged vestibular aqueduct with no problem at reimplantation). 2.4. Speech perception Mean scores on speech perception tests are shown in Table 3. The postoperative speech perception outcome measures showed no significant difference as compared to the best score achieved before reimplantation (phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores – Wilcoxon’s rank test for paired data – p > 0.05). The PBK performance improved by over 10% in 43.2% of children, was similar (an increase or a decrease in PBK
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
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Table 3 Speech discrimination was measured using phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores. Speech perception test scores 3 years after reimplantation, were slightly greater than the best results obtained with the first implant before reimplantation. However, no statistically significant difference could be found in PBK recognition test using no parametric tests for paired data (p = 0.08).
PBK (% correct score) Sentence recognition in quiet (% correct score) Sentence recognition in noise (SNR+ 10dB) (% correct score) CDT (speech tracking) (words correctly repeated per minute)
Best results with the first implant before reimplantation (mean)
Results one year after reimplantation (mean)
Results two years after reimplantation (mean)
Results three years after reimplantation (mean)
66% 78.9% 61.5%
61% 73% 57%
64% 75% 68%
70.6% 79.9% 70.1%
52
54
scores between the first and second implantation of less than 10%) in 40.5%, and showed a deterioration (decrease of more than 10%) in 16.2% (Fig. 1). 2.5. Angle of insertion analysis The mean insertion angle was 330.58 before reimplantation and 311.88 after (no statistical difference; p = 0.21). The PBK scores showed no statistical correlation with insertion angle values before or after reimplantation (respectively p = 0.35 and p = 0.65—ANOVA analysis). 2.6. Analysis of poor performances after reimplantation We classified 10 of the 45 reimplanted children, into a poor performance group due to their post-reimplantation PBK score of below 40%. We found partial insertion to be responsible for poor outcome in two children (20%—one meningitis and one genetic etiology), one (meningitis etiology) of whom suffered unexplained persistent pain leading to removal of the cochlear implant (nonuser). In three cases, neurologic disorders could explain the poor results (one severe CMV infection, one Landau–Klefner syndrome, and one cerebral palsy due to severe perinatal anoxia). We found no obvious explanation for the poor results obtained in four children (two with Usher type 1 syndrome, one with a GJB2 mutation, and one with unknown etiology). Among this poor performance group of 10 children, PBK scores were unchanged after reimplantation (scores under 40% before reimplantation) in
62
63.9
six children, and a deterioration of performance (decrease of more than 10% in PBK scores) occurred in three children (one child with a cerebral palsy, one with a partial insertion at reimplantation, and one without any obvious explanation for the deterioration). For ten of the reimplanted children in whom the best PBK score before reimplantation was below 40%, performance improved in four though not in six. 3. Discussion This study, concerned specifically with the pediatric population, reports a reimplantation rate of 8.3% (45 out of 539 children implanted). This rate falls within the range of values reported for children in the literature, from 4.5% [7] to 15.5% [4]. A greater reimplantation rate found among the pediatric population than in the adult population [1,3,4,6,7,14] is classically explained by the higher frequency of head trauma in children. Trauma accounted for only 8.7% of our cases of reimplantation, i.e. 0.7% of the 531 implanted children. In line with this, Farinetti and al. also found a lower rate of acquired failure due to trauma in children (less than 1% of their population [15]). Head trauma should not therefore be considered the principal cause of a greater rate of reimplantation in children. In our population, reimplantation for infection had the same prevalence as did reimplantation for trauma, The higher rate of reimplantation in the children of our series was primarly due to a malfunctioning device, found in 35 cases (77.7%), i.e. 6.5% of our pediatric-implanted population. This is in accordance with the literature that reports a rate of device failure ranging from 2.1%
Fig. 1. Open-set speech discrimination scores measured using phonetically balanced kindergarten (PBK) words at 3 years post-reimplantation were plotted as a function of PBK scores before reimplantation (in best conditions). The dashed line delimits the patients with similar performances before and after reimplantation. The bold line indicates the regression line.
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
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[16] to 10.6% [4]. Hard failure accounted for 4.45% of malfunctioning devices while soft failure accounted for 2.05%. The issue of device failure is controversial with some authors classifying certain soft failures as medical complications, leading to the variation of data among reports. Partial insertion during reimplantation surgery has been described, especially in meningitis or in otosclerosis [7]. Missing electrode rings at electrode array removal is uncommon and in the two cases of our series, did not jeopardize the complete insertion of the new electrode array during reimplantation. According to our results, cochlear reimplantation does not have a detrimental effect on auditory outcome, whatever the speech perception parameter used. Indeed, we found no influence of the device used for reimplantation, nor that of the angle of insertion at reimplantation, as reported previously [10]. While no statistical difference could be found, speech understanding performance in quiet improved in 43.2% of children in our series. On the other hand, only 16.2% of children showed a poorer performance after reimplantation. This has been reported by other authors: deterioration of outcomes in 2.9% of reimplantations for Rivas et al. in 2008 (adults [17]), in 37% of reimplantations for Henson et al. [5]. Poor post-reimplantation outcomes can be explained in some cases by a partial insertion; however, in most cases, no predictive factor can be found, as reflected in other studies [2,3,6]. Performance were not improved in three children with neurologic conditions: some neurologic disorders are not indication for reimplantation except for hard failure as reported by Blanchard et al. [11]. In our study, reimplantation improved the PBK score in four out of ten children, the best scores for whom were below 40% before reimplantation. Based on these findings, we believe that reimplantation can be considered in cases of unexplained poor outcome in implanted children. 4. Conclusion Cochlear implantation has become a routine procedure, leading in turn to an increase in the number of required reimplantations for medical/surgical complications or device malfunctions. The rate of reimplantation among our prelingually deafened pediatric population was 8.3%. In our experience, reimplantation is a safe surgical procedure with a complete insertion of the electrode array allowed in the vast majority of cases. Reimplantation had no significant effect on the average scores for speech perception outcome obtained in our population. We noted an improvement in PBK performance in 43.2%, no improvement in 40.5%, and a deterioration in 16.2% of children after reimplantation (explained in some by a partial insertion). Some children undergoing
reimplantation require a specific rehabilitation program to ensure they reap all its benefits.
References [1] J.T. Wang, A.Y. Wang, C. Psarros, M. Da Cruz, Rates of revision and device failure in cochlear implant surgery: a 30-year experience, Laryngoscope 124 (10) (2014) 2393–2399. [2] T.J. Balkany, A.V. Hodges, O. Go´mez-Marı´n, P.A. Bird, S. Dolan-Ash, S. Butts, et al., Cochlear reimplantation, Laryngoscope 109 (3) (1999) 351–355. [3] M. Coˆte´, P. Ferron, F. Bergeron, R. Bussie`res, Cochlear reimplantation: causes of failure, outcomes, and audiologic performance, Laryngoscope 117 (7) (2007) 1225–1235. [4] J. Gosepath, K. Lippert, A. Keilmann, W.J. Mann, Analysis of fifty-six cochlear implant device failures, ORL J. Otorhinolaryngol. Relat. Spec. 71 (3) (2009) 142–147. [5] A.M. Henson, W.H. Slattery, W.M. Luxford, D.M. Mills, Cochlear implant performance after reimplantation: a multicenter study, Am. J. Otol. 20 (1) (1999) 56–64. [6] L. Migirov, R. Taitelbaum-Swead, M. Hildesheimer, J. Kronenberg, Revision surgeries in cochlear implant patients: a review of 45 cases, Eur. Arch. Otorhinolaryngol. 264 (1) (2007) 3–7 (Off. J. Eur. Fed. Oto-Rhino-Laryngol. Soc. EUFOS Affil. Ger. Soc. Oto-Rhino-Laryngol.—Head Neck Surg.). [7] T. Sorrentino, M. Cote´, E. Eter, M.-L. Laborde, N. Cochard, O. Deguine, et al., Cochlear reimplantations: technical and surgical failures, Acta Otolaryngol. (Stockh) 129 (4) (2009) 380–384. [8] K.S. Van der Marel, J.J. Briaire, B.M. Verbist, R.M.S. Joemai, P-P.B.M. Boermans, F.A.W. Peek, et al., Cochlear reimplantation with same device: surgical and audiologic results, Laryngoscope 121 (7) (2011) 1517–1524. [9] J. Sunde, J.B. Webb, P.C. Moore, M.B. Gluth, J.L. Dornhoffer, Cochlear implant failure, revision, and reimplantation, Otol. Neurotol. 34 (Dec (9)) (2013) 1670–1674 (Off. Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol.). [10] S. Mahtani, F. Glynn, D.J. Mawman, M.P. O’Driscoll, K. Green, I. Bruce, et al., Outcomes of cochlear reimplantation in adults, Otol. Neurotol. 35 (8) (2014) 1366–1372 (Off. Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol.). [11] M. Blanchard, B. Thierry, F. Glynn, A. De Lamaze, E.N. Garabe´dian, N. Loundon, Cochlear implant failure and revision surgery in pediatric population, Ann. Otol. Rhinol. Laryngol. 124 (3) (2015) 227–231. [12] Y. Olgun, A.F. Bayrak, T. Catli, M.E. Ceylan, R. Aydin, U. Duzenli, et al., Pediatric cochlear implant revision surgery and reimplantation: an analysis of 957 cases, Int. J. Pediatr. Otorhinolaryngol. 78 (10) (2014) 1642–1647. [13] B.M. Verbist, M.W. Skinner, L.T. Cohen, P.A. Leake, C. James, C. Boe¨x, et al., Consensus panel on a cochlear coordinate system applicable in histologic, physiologic, and radiologic studies of the human cochlea, Otol. Neurotol. 31 (Jul (5)) (2010) 722–730 (Off. Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol.). [14] K.D. Brown, S.S. Connell, T.J. Balkany, A.E. Eshraghi, F.F. Telischi, S.A. Angeli, Incidence and indications for revision cochlear implant surgery in adults and children, Laryngoscope 119 (1) (2009) 152–157. [15] A. Farinetti, D. Ben Gharbia, J. Mancini, S. Roman, R. Nicollas, J.-M. Triglia, Cochlear implant complications in 403 patients: comparative study of adults and children and review of the literature, Eur. Ann. Otorhinolaryngol. Head Neck Dis. 131 (Jun (3)) (2014) 177–182. ¨ . Tarkan, U ¨ zdemir, O ¨ . Su¨rmeliog˘lu, F. C¸etik, M. Kırog˘lu, et al., Surgical ¨ . Tuncer, S. O [16] O and medical management for complications in 475 consecutive pediatric cochlear implantations, Int. J. Pediatr. Otorhinolaryngol. 77 (4) (2013) 473–479. [17] A. Rivas, A.L. Marlowe, J.E. Chinnici, J.K. Niparko, H.W. Francis, Revision cochlear implantation surgery in adults: indications and results, Otol. Neurotol. 29 (Aug (5)) (2008) 639–648 (Off. Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol.).
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
PEDOT-7511; No. of Pages 4 International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Outcomes after cochlear reimplantation in children Franc¸oise Sterkers, Fanny Merklen, Jean Pierre Piron, Adrienne Vieu, Fre´de´ric Venail, Alain Uziel, Michel Mondain * Cochlear Implant Center Montpellier-Palavas, University Hospital of Montpellier, 34295 Montpellier Cedex 5, France
A R T I C L E I N F O
A B S T R A C T
Article history: Received 26 November 2014 Received in revised form 16 February 2015 Accepted 17 March 2015 Available online xxx
Objectives: With cochlear implantation now a routine procedure, reimplantation is becoming more commonplace for medical/surgical complications or device malfunctions. This study investigated the indications for reimplantation and the auditory outcomes following reimplantation surgery in prelingually-deafened children. Methods: Of the 539 prelingually deafened children implanted between 1990 and 2013, 45 were reimplanted (8.3% of implantations). Causes of reimplantation, type of device and angle of insertion at initial implantation were recorded, as well as type of implant reinserted, number of electrodes inserted and angle of insertion (calculated on cone beam computed tomography) on reimplantation, and finally any surgical findings. Speech perception test scores (phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores) were obtained 1, 2 and 3 years after reimplantation, and compared against the best speech recognition score obtained with the first implant before failure. Results: Medical reasons for reimplantation were found in 10 cases (22.2%). A malfunctioning device had occurred in 35 cases (77.7%) including hard failure in 24 and soft failure in 11. Complete insertion was achieved in the scala tympani in 42 cases and in the scala vestibuli in one case; partial insertion occurred in the remaining two cases. In two cases, one or two electrode rings snatched off from the electrode array during removal. The mean insertion angle was 330.58 before surgery and 311.88 after reimplantation (no statistical difference p = 0.48). The postoperative speech perception outcome measures showed no significant difference to the best score before reimplantation. Angle of insertion, type of device and etiology of deafness did not influence the results. The PBK performance improved over 10% in 43.2% of children, was similar in 40.5%, and showed a more than 10% decrease in 16.2% of children after reimplantation. The latter decline in performance was explained for some children by a partial insertion. Conclusions: Reimplantation has no negative effect on auditory outcome. In rare cases, speech perception outcome may not improve, requiring a specific rehabilitation program. ß 2015 Elsevier Ireland Ltd. All rights reserved.
Keywords: Cochlear implant Child Reimplantation Device failure
Cochlear implantation has become a routine procedure for restoring serviceable hearing to children having suffered a bilateral severe-to-profound sensorineural hearing loss with little or no benefit from hearing aids. In some cases, a reimplantation surgery is mandatory. The reasons for cochlear implant explantation or reimplantation can be divided into medical or surgical complications and device malfunctions. Device malfunctions comprise hard
* Corresponding author. Tel.: +33 467336804. E-mail addresses: [email protected], [email protected] (M. Mondain).
and soft failures and account for the majority of reimplantation surgeries. Several studies have investigated the indications, complications and results of reimplantation [1–10]. Most of these included both adults and children and showed no detrimental effect on speech perception outcome of reimplantation. Few specific pediatric series has been published recently [11,12], yet children may have a slightly higher rate of reimplantation, believed due to a higher incidence of trauma among children than in adults. This study aimed to investigate the indications for reimplantation, to assess the auditory outcomes following reimplantation surgery in prelingually deafened children, and to identify factors that may influence these outcomes.
http://dx.doi.org/10.1016/j.ijporl.2015.03.015 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
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PEDOT-7511; No. of Pages 4 F. Sterkers et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
2
1. Materials and methods Medical reports from a total of 539 prelingually deafened children implanted between 1990 and 2013 were retrospectively analyzed. Overall, 45 cochlear implants had been replaced (8.3% of the implantations). Age at initial implantation ranged from 1.1 to 14.9 years (mean 5.07 years). Twenty boys and 25 girls aged from 5.2 to 22.8 years (average 13.9 years) underwent a reimplantation. Time from initial implantation to reimplantation ranged from 5 months to 21.7 years. Etiologies, detailed in Table 1, were primarily genetics-related (29%), meningitis (13%), and unknown (31%). Medical records were reviewed to identify causes of reimplantation, type of device and length of insertion at initial implantation, type of implant reinserted, number of electrodes inserted and angle of insertion (calculated on cone beam computed tomography [13]) on reimplantation, and surgical findings. The primary outcome measures were speech discrimination measured using phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores. Speech perception test scores were obtained 1, 2 and 3 years after reimplantation, and compared to the best results obtained with the first implant before failure. For individual patients, an increase in PBK scores between the first and second implantation of 10% or greater was regarded as an improved performance. Similarly, a reduction in PKB scores of 10% or more was regarded as a deterioration in performance. Statview software (Abacus1) for PC was used for statistical analysis. Statistical differences were analyzed using a non parametric test for paired data (Wilcoxon’s rank test). Statistical significance was set at the 5% level. Correlations were analyzed using ANOVA analysis.
2. Results 2.1. Type of device Three manufacturers were used for implantation: Cochlear, Neurelec and Advanced Bionics. Table 2 indicates the specific distribution of cochlear implant models (Cochlear 80% of population, Neurelec 4.5%, AB 15.5%) that required reimplantation and that of the models used to replace them. Forty-four reimplantations were performed with a device from the same company. The vast majority of Nucleus CI 22 were upgraded with a Nucleus CI 24 device, while the vast majority of the Clarion 1.2 CI were upgraded with a Clarion HF 90 CI. Table 1 Etiology of deafness for the 45 reimplanted children. N
Etiology
%
Genetics—nonsyndromic
GJB2 or GJB6 gene mutation Other
4 9
9 20
Genetics—syndromic
Usher type 1 Waardenburg Down syndrome Landau–Kleffner Mohr–Tranebjaerg syndrome
5 1 1 1 1
11 2 2 2 2
Meningitis Labyrinthitis CMV
6 1 1
13 2 2
1
2
14
31
Infectious
Perinatal Anoxia Unknown
Table 2 Distribution of the models of cochlear implants at implantation and at reimplantation. CI model
At implantation
At reimplantation
Nucleus CI22 Nucleus CI24 (M–R–RE) Nucleus CI512 Clarion 1.2 Clarion HF 90 Digisonic
24 10 2 6 1 2
3 33 1 0 6 2
2.2. Reasons for reimplantation Medical reasons accounted for the reimplantation in 10 cases (22.2%). These were infection in four cases all related to flap problems, head trauma with decreasing performance in another four, and neurologic problems in two. One of these latter children had Landau–Kleffner syndrome without improvement of language skills despite a 20% correct score on open-set recognition tests, and the other child required deep brain stimulation to control a severe dystonia (Mohr–Tranebjaerg syndrome). Malfunctioning device accounted for the decrement in clinical performance found in 35 cases (77.7%). This was a hard failure (abnormal integrity testing) in 24 cases (53.3%) and a soft failure (normal integrity testing though poor performance from the implant and/or nonauditory symptoms) in the remaining 11 (24.4%). 2.3. Surgical findings All reimplantations were performed at the same side, with one patient undergoing bilateral implantation during the reimplantation surgery. We achieved complete insertion in the scala tympani in 42 cases (93.3%). Partial insertion occurred in two cases: one child with unknown etiology of deafness with partial insertion at reimplantation (Nucleus CI24M; 12 active electrodes outside the cochleostomy), and the other child deafened from meningitis in whom insertion was partial during both implantation and reimplantation (Nucleus CI 512; 6 active electrodes outside the cochleostomy at reimplantation–ossification of the basal turn with partial insertion at implantation). Reimplantation was easy in all other cases of post-meningitis deafness, even in a child with round window ossification at implantation. In one case, we performed a complete insertion in the scala vestibuli at reimplantation. In two cases, electrode rings were snatched off the electrode array during removal and were left in place (one apical electrode ring snatched off a Digisonic SP device in one child, and two apical electrodes in another implanted with a Nucleus CI22 device): complete insertion of the new electrode array was achieved in both cases. Three cases required a complementary tympanoplasty (one eardrum perforation–two tympanic membrane retractions). No reimplantation was performed in cochlear abnormality cases (two children has an enlarged vestibular aqueduct with no problem at reimplantation). 2.4. Speech perception Mean scores on speech perception tests are shown in Table 3. The postoperative speech perception outcome measures showed no significant difference as compared to the best score achieved before reimplantation (phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores – Wilcoxon’s rank test for paired data – p > 0.05). The PBK performance improved by over 10% in 43.2% of children, was similar (an increase or a decrease in PBK
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
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Table 3 Speech discrimination was measured using phonetically balanced kindergarten (PBK) words, open-set sentence testing in quiet and in noise (S/N+ 10dB SNR), and speech tracking scores. Speech perception test scores 3 years after reimplantation, were slightly greater than the best results obtained with the first implant before reimplantation. However, no statistically significant difference could be found in PBK recognition test using no parametric tests for paired data (p = 0.08).
PBK (% correct score) Sentence recognition in quiet (% correct score) Sentence recognition in noise (SNR+ 10dB) (% correct score) CDT (speech tracking) (words correctly repeated per minute)
Best results with the first implant before reimplantation (mean)
Results one year after reimplantation (mean)
Results two years after reimplantation (mean)
Results three years after reimplantation (mean)
66% 78.9% 61.5%
61% 73% 57%
64% 75% 68%
70.6% 79.9% 70.1%
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scores between the first and second implantation of less than 10%) in 40.5%, and showed a deterioration (decrease of more than 10%) in 16.2% (Fig. 1). 2.5. Angle of insertion analysis The mean insertion angle was 330.58 before reimplantation and 311.88 after (no statistical difference; p = 0.21). The PBK scores showed no statistical correlation with insertion angle values before or after reimplantation (respectively p = 0.35 and p = 0.65—ANOVA analysis). 2.6. Analysis of poor performances after reimplantation We classified 10 of the 45 reimplanted children, into a poor performance group due to their post-reimplantation PBK score of below 40%. We found partial insertion to be responsible for poor outcome in two children (20%—one meningitis and one genetic etiology), one (meningitis etiology) of whom suffered unexplained persistent pain leading to removal of the cochlear implant (nonuser). In three cases, neurologic disorders could explain the poor results (one severe CMV infection, one Landau–Klefner syndrome, and one cerebral palsy due to severe perinatal anoxia). We found no obvious explanation for the poor results obtained in four children (two with Usher type 1 syndrome, one with a GJB2 mutation, and one with unknown etiology). Among this poor performance group of 10 children, PBK scores were unchanged after reimplantation (scores under 40% before reimplantation) in
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six children, and a deterioration of performance (decrease of more than 10% in PBK scores) occurred in three children (one child with a cerebral palsy, one with a partial insertion at reimplantation, and one without any obvious explanation for the deterioration). For ten of the reimplanted children in whom the best PBK score before reimplantation was below 40%, performance improved in four though not in six. 3. Discussion This study, concerned specifically with the pediatric population, reports a reimplantation rate of 8.3% (45 out of 539 children implanted). This rate falls within the range of values reported for children in the literature, from 4.5% [7] to 15.5% [4]. A greater reimplantation rate found among the pediatric population than in the adult population [1,3,4,6,7,14] is classically explained by the higher frequency of head trauma in children. Trauma accounted for only 8.7% of our cases of reimplantation, i.e. 0.7% of the 531 implanted children. In line with this, Farinetti and al. also found a lower rate of acquired failure due to trauma in children (less than 1% of their population [15]). Head trauma should not therefore be considered the principal cause of a greater rate of reimplantation in children. In our population, reimplantation for infection had the same prevalence as did reimplantation for trauma, The higher rate of reimplantation in the children of our series was primarly due to a malfunctioning device, found in 35 cases (77.7%), i.e. 6.5% of our pediatric-implanted population. This is in accordance with the literature that reports a rate of device failure ranging from 2.1%
Fig. 1. Open-set speech discrimination scores measured using phonetically balanced kindergarten (PBK) words at 3 years post-reimplantation were plotted as a function of PBK scores before reimplantation (in best conditions). The dashed line delimits the patients with similar performances before and after reimplantation. The bold line indicates the regression line.
Please cite this article in press as: F. Sterkers, et al., Outcomes after cochlear reimplantation in children, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.03.015
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[16] to 10.6% [4]. Hard failure accounted for 4.45% of malfunctioning devices while soft failure accounted for 2.05%. The issue of device failure is controversial with some authors classifying certain soft failures as medical complications, leading to the variation of data among reports. Partial insertion during reimplantation surgery has been described, especially in meningitis or in otosclerosis [7]. Missing electrode rings at electrode array removal is uncommon and in the two cases of our series, did not jeopardize the complete insertion of the new electrode array during reimplantation. According to our results, cochlear reimplantation does not have a detrimental effect on auditory outcome, whatever the speech perception parameter used. Indeed, we found no influence of the device used for reimplantation, nor that of the angle of insertion at reimplantation, as reported previously [10]. While no statistical difference could be found, speech understanding performance in quiet improved in 43.2% of children in our series. On the other hand, only 16.2% of children showed a poorer performance after reimplantation. This has been reported by other authors: deterioration of outcomes in 2.9% of reimplantations for Rivas et al. in 2008 (adults [17]), in 37% of reimplantations for Henson et al. [5]. Poor post-reimplantation outcomes can be explained in some cases by a partial insertion; however, in most cases, no predictive factor can be found, as reflected in other studies [2,3,6]. Performance were not improved in three children with neurologic conditions: some neurologic disorders are not indication for reimplantation except for hard failure as reported by Blanchard et al. [11]. In our study, reimplantation improved the PBK score in four out of ten children, the best scores for whom were below 40% before reimplantation. Based on these findings, we believe that reimplantation can be considered in cases of unexplained poor outcome in implanted children. 4. Conclusion Cochlear implantation has become a routine procedure, leading in turn to an increase in the number of required reimplantations for medical/surgical complications or device malfunctions. The rate of reimplantation among our prelingually deafened pediatric population was 8.3%. In our experience, reimplantation is a safe surgical procedure with a complete insertion of the electrode array allowed in the vast majority of cases. Reimplantation had no significant effect on the average scores for speech perception outcome obtained in our population. We noted an improvement in PBK performance in 43.2%, no improvement in 40.5%, and a deterioration in 16.2% of children after reimplantation (explained in some by a partial insertion). Some children undergoing
reimplantation require a specific rehabilitation program to ensure they reap all its benefits.
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