- Email: [email protected]
Hearing improvement with softband and implanted bone-anchored hearing devices and modified implantation surgery in patients with bilateral microtia-atresia
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Hearing improvement with softband and implanted bone-anchored hearing devices and modified implantation surgery in patients with bilateral microtia-atresia
T
Yibei Wang, Xinmiao Fan, Pu Wang, Yue Fan, Xiaowei Chen∗ Department of Otolaryngology, Peking Union Medical College Hospital, Beijing, 100730, China
A R T I C L E I N F O
A B S T R A C T
Keywords: Bilateral microtia-atresia Bone anchored hearing devices Auditory development Modified implantation surgery Hearing improvement Speech discrimination
Objective: To evaluate auditory development and hearing improvement in patients with bilateral microtiaatresia using softband and implanted bone-anchored hearing devices and to modify the implantation surgery. Methods: The subjects were divided into two groups: the softband group (40 infants, 3 months to 2 years old, Ponto softband) and the implanted group (6 patients, 6–28 years old, Ponto). The Infant-Toddler Meaning Auditory Integration Scale was used conducted to evaluate auditory development at baseline and after 3, 6, 12, and 24 months, and visual reinforcement audiometry was used to assess the auditory threshold in the softband group. In the implanted group, bone-anchored hearing devices were implanted combined with the auricular reconstruction surgery, and high-resolution CT was used to assess the deformity preoperatively. Auditory threshold and speech discrimination scores of the patients with implants were measured under the unaided, softband, and implanted conditions. Results: Total Infant-Toddler Meaning Auditory Integration Scale scores in the softband group improved significantly and approached normal levels. The average visual reinforcement audiometry values under the unaided and softband conditions were 76.75 ± 6.05 dB HL and 32.25 ± 6.20 dB HL (P < 0.01), respectively. In the implanted group, the auditory thresholds under the unaided, softband, and implanted conditions were 59.17 ± 3.76 dB HL, 32.5 ± 2.74 dB HL, and 17.5 ± 5.24 dB HL (P < 0.01), respectively. The respective speech discrimination scores were 23.33 ± 14.72%, 77.17 ± 6.46%, and 96.50 ± 2.66% (P < 0.01). Conclusions: Using softband bone-anchored hearing devices is effective for auditory development and hearing improvement in infants with bilateral microtia-atresia. Wearing softband bone-anchored hearing devices before auricle reconstruction and combining bone-anchored hearing device implantation with auricular reconstruction surgery may bethe optimal clinical choice for these patients, and results in more significant hearing improvement and minimal surgical and anesthetic injury.
1. Introduction Congenital microtia-atresia results from developmental defects of the auricle and is generally associated with malformations of the external auditory canal and the middle ear. The incidence of congenital microtia-atresia is estimated to be between 0.81 in 10,000 and 3.06 in 20,000 live births in China [1]. Approximately 10% of patients with congenital microtia-atresia have the condition in both ears. Patients with congenital microtia-atresia often experience conductive hearing loss with an air-bone gap of 50–60 dB due to bony or soft-tissue occlusion [2]. The first two years of life are critical in language acquisition. During
this period, hearing ability is crucial in learning spoken language. There is growing evidence that the absence of sound and language access leads to cognitive delay, mental health difficulties, a higher possibility of traumatic accidents, and limited literacy [3]. Early intervention for children with hearing impairment is thus essential. Patients with microtia-atresia have lower self-esteem and social dysfunction [4], and require hearing rehabilitation and auricular reconstruction surgery to improve their appearance. Until now, the use of bone-anchored hearing devices has been one of the most reliable methods for auditory rehabilitation [5,6]. Softband bone-anchored hearing devices fix the processor around the head via a soft band. Sound is thus transmitted directly by the skull bone to the
∗
Corresponding author. Department of Otolaryngology, Peking Union Medical College Hospital, #1 shuaifuyuan, Beijing, 100730, China. E-mail addresses: [email protected] (Y. Wang), [email protected] (X. Fan), [email protected] (P. Wang), [email protected] (Y. Fan), [email protected] (X. Chen). https://doi.org/10.1016/j.ijporl.2017.11.010 Received 27 September 2017; Received in revised form 30 October 2017; Accepted 1 November 2017 Available online 14 November 2017 0165-5876/ © 2017 Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
includes 10 items grouped into 3 domains: vocalization behavior (items 1 and 2), alerting to sounds (items 3–6), and deriving meaning from sound (items 7–10). Each question has five different response alternatives: 0 = never, 1 = rarely, 2 = occasionally, 3 = frequently, and 4 = always [11]. The Infant-Toddler Meaningful Auditory Integration Scale was used to evaluate auditory development at baseline (0 months), and 6, 12, and 24 months after wearing the Ponto softband. Visual reinforcement audiometry was used to assess the auditory threshold after wearing the Ponto softband for 6 months under unaided and aided conditions.
inner ear. Implanted bone-anchored hearing devices transmit sound using a titanium implant, which couples the vibration transducer to the skull bone via a skin-penetrating abutment [7]. Operation timing and the position of implanted bone-anchored hearing devices are crucial. Our group has previously studied hearing improvement following the use of softband and implanted bone-anchored hearing devices in patients with bilateral microtia-atresia [8]. We have also recommended the early use of softband bone-anchored hearing devices in infants with bilateral microtia-atresia [9]. The growth curve describing auditory development in infants with bilateral microtia-atresia using softband bone-anchored hearing devices is yet unknown. Choosing the operation time and implantation position are also major challenges for surgeons. Here we aimed to evaluate the auditory development of Mandarinspeaking children with bilateral microtia-atresia using the Ponto softband by following up 40 infants over two years. We also aimed to modify the implantation surgery by combining it with auricle reconstruction.
2.2.2. Implanted group 2.2.2.1. Imaging evaluation and modified implantation surgery. All patients underwent preoperative temporal bone high-resolution computed tomography to evaluate temporal bone thickness and the middle ear structures. Patients were graded using the Jahrsdoerfer grading scale. According to Jahrsdoerfer criteria, 6 patients who scored less than 6 points in both ears were considered poor candidates for hearing reconstruction. Therefore, atresiaplasty was not performed in these patients. In the first stage of auricular reconstruction surgery, a soft-tissue skin expander was implanted in the mastoid region to enable us to obtain a sufficient amount of skin. In the second stage, an autogenous rib cartilage framework was carved to reconstruct the auricle. The third stage comprised the second revision of the auricular reconstruction. Ponto implantation was performed during the auricular reconstructive surgery, especially the second stage. In patients who had undergone the second stage of surgery, we were able to combine Ponto implantation with the third stage. In patients who required 2-stage implantation, the titanium implant was inserted into the mastoid bone at the end of the soft-tissue skin expander or the auricle reconstruction sugeries. The abutment was attached to the titanium screw during the next stage of auricle reconstructions, which was performed 4–6 months later.
2. Methods 2.1. Subjects This single center retrospective study included 46 patients with bilateral microtia-atresia who were enrolled at Peking Union Medical College Hospital in Beijing, China, from January 1, 2014, to April 30, 2016. The study subjects comprised 40 infants (aged 3 months to 2 years) and 6 patients with implants (aged 6–28 years). Ethical approval for this study was obtained from the Institutional Review Board of Peking Union Medical College Hospital. Parents of all patients in the study provided written informed consent for the use of soft-band and implanted bone-anchored hearing devices and the tests. 2.1.1. Softband group The softband group consisted of 40 infants with bilateral microtiaatresia comprising 23 boys and 17 girls ranging in age from 3 months to 2 years (average age, 8.45 ± 5.67 months; median age, 7 months), wearing Ponto softband (Ponto pro, made by Oticon). All 40 patients were classified as grade III according to the Marx classification of the severity of deformity and bilateral aural atresia [10]. The average bone conduction threshold of the deformed ear, as assessed using the auditory brainstem response, was 18.13 ± 4.84 dB nHL (range, 0–25 dB nHL) and the average air conduction threshold was 76.38 ± 4.53 dB nHL (range, 70–85 dB nHL).
2.2.2.2. Audiological evaluation. Sound field pure-tone audiometry was conducted for all patients at 250, 500, 1,000, 2,000, 4,000, and 8000 Hz under unaided, softband-aided, and implantation-aided conditions. The speech perception tests were conducted in a sound field using the Mandarin speech test system [12]. A bisyllabic test list was used to obtain speech discrimination scores under the unaided, softband, and Ponto-implanted conditions. All test materials were presented twice. The average score was used in the final analysis. 2.3. Statistical analysis
2.1.2. Implanted group Six patients aged 6–28 years (average age, 15.17 ± 10.00 years) received Ponto (Ponto, made by Oticon) implantation surgery between May 2015 and December 2016 after using the Ponto softband (Ponto pro, made by Oticon) for 3–12 months (mean, 6 months). All 6 patients, who were male, were classified as grade III according to the Marx classification of the severity of deformity and bilateral aural atresia [10]. The average bone conduction threshold of the deformed ear, as assessed using pure tone audiometry, was 17.50 ± 2.74 dB HL (range, 0–20 dB HL), and the average air conduction threshold was 76.67 ± 5.16 dB HL (range, 70–85 dB HL) for tones between 250 and 8000 Hz. One patient had undergone auricle reconstruction surgery in 1992 in another hospital. This patient underwent bilateral bone-anchored hearing device implantation.
All data were analyzed using SPSS (version 19.0, IBM Corp.). Continuous variables are presented as mean ± SD. Infant-Toddler Meaningful Auditory Integration Scale scores were compared to normal scores for this scale [11]. Repeated measures ANOVAs with Greenhouse-Geisser corrections were used to assess differences between the standard and softband-aided Infant-Toddler Meaningful Auditory Integration Scale scores. Paired t tests were used to assess the differences between patients with and without Ponto softbands in the softband group. ANOVAs were used to assess differences among the unaided, softband-aided, and implant-aided conditions in the implanted group. P values < 0.01 were considered statistically significant. 3. Results
2.2. Main outcome measures
3.1. Auditory development in infants using Ponto softbands
2.2.1. Softband group Auditory and speech development of infants were mainly assessed using the translated Chinese version of the Infant-Toddler Meaningful Auditory Integration Scale. The Infant-Toddler Meaningful Auditory Integration Scale is a structured parent interview questionnaire that
The Infant-Toddler Meaningful Auditory Integration Scale scores of the 40 children in the softband group were assessed at baseline (0 month), and 3, 6, 12, and 24 months after baseline. The mean follow-up time was 21.9 ± 4.6 months. 17.5% of the patients were lost to followup. The patients wore the devices for an average of 6.57 ± 1.22 h per 121
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
Fig. 1. IT-MAIS Total Score In this figure, the upper red curve refers to the scores of normal level, while the broken lines referringreferring to the scores of 40 microtia-atresia infants. ITMAIS total scores of these infants were lower than the standard, which were found to have improved significantly and approaching the normal level gradually with time after wearing the softband Ponto.For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.
3.3. Hearing improvement
day. As shown in Fig. 1, Infant-Toddler Meaningful Auditory Integration Scale total scores in the softband group were lower than the standard scores [11], although they improved significantly and approached normal levels gradually with time. The average difference in InfantToddler Meaningful Auditory Integration Scale scores of the patients in the softband group and standard scores at baseline (0 months), and 3, 6, 12, and 24 months after baseline were 44.30 ± 15.31%, 38.15 ± 12.87%, 29.18 ± 10.95%, 19.80 ± 10.55% and 5.24 ± 11.55%, respectively. The difference was greatest at baseline and decreased gradually with time. This change was statistically significant (Greenhouse-Geisser, F = 144.09, P < 0.001) (Fig. 2).
3.3.1. Hearing threshold In the softband group, the average unaided hearing thresholds measured using visual reinforcement audiometry were 76.75 ± 6.05 dB HL, while the average hearing thresholds when using a Ponto softband were 32.26 ± 6.20 dB HL. There was a statistically significant improvement of 44.50 ± 3.89 dB HL in the threshold, as assessed using a paired t-test (t = 72.36, P < 0.01). In the implanted group, the mean preoperative sound field threshold was 59.17 ± 3.76 dB HL in patients with unaided hearing. The mean softband and postoperative sound field thresholds were 32.5 ± 2.74 dB HL, and 17.5 ± 5.24, respectively. These values were significantly different (F = 163.05, P < 0.001). The Ponto softband and the implantation procedure resulted in mean hearing improvements of 26.67 ± 4.08 dB and 41.67 ± 5.16 dB. The implanted Ponto resulted in an improvement greater than that achieved using the softband by 15 ± 4.72 dB, as shown in http://jamanetwork.com/ journals/jamaotolaryngology/fullarticle/1816004 Fig. 5.
3.2. Modified implantation surgery The average Jahrsdoerfer grading scale score was 4.5 ± 0.55. All patients had stapes abnormalities. Of the 6 patients in the group, 5 had undergone Ponto implantation combined with auricular reconstructive surgery. One patient who already had reconstructed auricles underwent bilateral implantation in one surgery using the conventional surgical method. Among the patients in this group, 2 children younger than 7 years of age required 2-stage surgeries for implantation. In one of these children, we inserted the titanium implant at the end of the soft-tissue skin expander surgery and attached the abutment at the end of the auricular reconstruction surgery (Fig. 3). In the other child, we inserted the titanium implant at the end of the second stage of the auricular reconstruction surgery and attached the abutment at the end of the third stage of the surgery. For the other 3 patients, we inserted the titanium implant and attach it to the abutment at the end of the second stage of the auricular reconstruction surgery (Fig. 4). At this time, no skin necrosis and or traumatic extrusions have been reported.
3.3.2. Speech discrimination score In the implanted group, the mean unaided speech discrimination score, as evaluated using the bisyllabic test (presentation level, 65 dB HL) was 23.33 ± 14.72%. The mean score was 77.17 ± 6.46% in patients using the Ponto softband and 96.50 ± 2.66%in patients with implanted Ponto devices. These improvements of 53.84 ± 15.29% and 73.17 ± 15.29% were statistically significant (F = 97.45, P < 0.001). Ponto implantation led to a greater improvement 19.33 ± 4.89% than the softband, as shown in http://jamanetwork. com/journals/jamaotolaryngology/fullarticle/1816004 Fig. 6. 122
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
Fig. 2. The difference of the IT-MAIS Scores Fig. 2 showed the differences of the IT-MAIS scores compared to the standard scores after wearing the softband Ponto for baseline (0 months), three months, six months, twelve months and twenty-four months. The difference was greatest in baseline and decrease gradually with time, which is statistically significant (Greenhouse-Geisser, F = 144.09,P < 0.001).
4. Discussion
bone-anchored hearing devices in a two-year follow-up study of 40 infants. We found that the softband bone-anchored hearing device had effective impacts on auditory development. Using questionnaires completed by the patient's parents is the best way to assess early prelingual auditory development and the efficacy of hearing intervention, as younger children have difficulty performing other types of tests used to assess auditory development. The InfantToddler Meaningful Auditory Integration Scale has been reported to have good reliability and validity in measuring auditory development and hearing rehabilitation efficacy [16]. A study of 120 infants and toddlers with normal hearing born to Mandarin-speaking parents was used to establish normal scores for the spontaneous detection, recognition, and discrimination of sounds. This study provides us with a trajectory for standard Infant-Toddler Meaningful Auditory Integration Scale total scores during the first two years of life [11]. We found that infants with bilateral microtia-atresia had deficits in auditory development, with a median decrease of 44.30 ± 15.31% in the Infant-Toddler Meaningful Auditory Integration Scale total score. This finding reflects the necessity for early intervention to improve hearing in infants with bilateral microtia-atresia. The current treatment options for bilateral microtia-atresia are varied but limited. They include surgical reconstruction of the external auditory canal and boneanchored hearing device implantation. Bone-anchored hearing device
4.1. The efficacy and superiority of softband bone-anchored hearing devices in children with bilateral microtia-atresia Hearing plays a crucial role in the early stages of language development. Inadequate access to spoken language may delay the development of neuro-linguistic structures of the brain, and especially grammar and second language acquisition [13]. The severe conductive hearing loss associated with bilateral microtia-atresia gives rise to communication barriers and delays in speech-language abilities. Recent evidence suggests that children with microtia-atresia are at risk for delays in speech-language development and cognition, and behavioral problems that can affect functioning in school [14]. Additionally, development of the central auditory system might be altered due to the limited stimulation caused by the significant air-bone gap in these patients [15]. There is consensus that early hearing rehabilitation contributes to auditory and language development. To our knowledge, there are yet no studies incorporating a large sample and follow-up observation to investigate auditory development in patients with bilateral microtia-atresia using the Ponto softband bone-anchored hearing device. We aimed to evaluate auditory development in Mandarin-speaking patients with bilateral microtia-atresia using softband
Fig. 3. The modified 2-stage Ponto implantation surgery Fig. 3 showed the child who was inserted the titanium implant at the end of the soft-tissue skin expander surgery and attached the abutment at the end of the second stage of the auricular reconstruction surgery. A and B showed the titanium implant which was inserted in the soft –tissue skin expender surgery. C showed the attachment of the abutment.
123
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
Fig. 4. The modified implantation surgery Fig. 4 showed the Ponto implantation surgery combined with the second stage of the auricular reconstruction surgery. We inserted the titanium implant and attach it to the abutment after the auricle was settled, as shown in A. The skin above the abutment was eliminated by skin punch to make the minimal injury as shown in B. The healing cap was used to reduce the infection as shown in C.
Fig. 5. Hearing Thresholds of the Implanted Group Fig. 5 showed the mean hearing thresholds in the implanted group under the unaided, softband and implanted conditions. The soft-band Ponto and the implantation gave a mean hearing improvement of 26.67 ± 4.08 dB and 41.67 ± 5.16 dB, which was statistically significant (P < 0.001).
Fig. 6. The Speech Discrimination Score of the Implanted Group Fig. 6 showed the mean speech discrimination in the implanted group under the unaided, softband and implanted conditions. The softband and the implantation gave a mean hearing improvement of 53.84 ± 15.29% and 73.17 ± 15.29%, which was statistically significant (P < 0.001).
implantation and aural reconstruction surgeries certainly improve speech recognition ability [17–23]. It is undeniable that there are many postoperative complications following aural reconstruction surgery, such as facial nerve injury and meatal stenosis [24,25]. In patients with microtia-atresia, aural reconstruction surgery may lead to deficits in the blood supply of the flap used in the secondary stage of auricle reconstruction surgery. This may in turn increase surgical difficulties and complications postoperatively. The use of softband bone-anchored hearing devices is a common clinical choice for the treatment of infants with bilateral microtiaatresia. These devices are used as early as 3 months of age, before the patient's skull bones are sufficiently thick for implantation surgery. After wearing the softband bone-anchored hearing device for 6 months, most patients experienced improved auditory development, and the developmental trajectories of the patients gradually neared or reached those of healthy infants and children. This finding is comparable to that of our previous study [26]. In the current study, we further characterized the auditory developmental trajectories of infants with bilateral microtia-atresia wearing softband bone-anchored hearing devices. Auditory development is encouraging in these patients. The hearing threshold was 32.26 ± 6.20 dBHL, as tested using visual reinforcement audiometry, which is an improvement of 44.50 ± 3.89 dB.
Considering the demand for hearing improvement and the surgical complications described above, we recommend the use of softband bone-anchored hearing devices to improve hearing ability before auricular reconstruction surgery becomes the optimal treatment choice in children with bilateral microtia-atresia.
4.2. Modified surgery combining bone-anchored hearing device implantation with auricle reconstruction Auricular reconstruction surgery using autologous cartilage is currently the most effective method used to correct deformities due to microtia [27]. This procedure results in both aesthetic and psychosocial improvements. As discussed above, hearing rehabilitation is essential in patients with bilateral microtia-atresia [28]. Ponto implantation was performed at the end of the surgery after the auricle was settled. The position of the screw should be carefully determined to prevent the sound processor from impeding the auricular reconstruction and impairing the blood supply of the flap. We always settle the screw 5–7 cm away from the tragus of the reconstructed auricle at a 45-degree angle. We do not remove subcutaneous tissue during this procedure. We use a punch to remove the skin above the screw in order to establish contact with the abutment. Combining Ponto implantation with the second 124
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
administered the IT-MAIS questionnaire and collected the data. YBW and PW tested the hearing thresholds and assigned speech discrimination scores. YBW analyzed the data. YBW wrote the paper. XWC and YF revised the paper critically for important intellectual content. All authors read and approved the final manuscript.
stage of ear reconstruction helps position the screw and can spare the patients from additional incision and anesthetic use. Some young children require 2-stage implantation because the thinner and less dense skull bone requires a period of 4–6 months for osseointegration of the fixture. In this study, 2 patient younger than 7 years of age underwent 2-stage implantation. Combining the 2 stages of the implantation with 2 steps of the auricular reconstruction surgery reduced the total time of treatment. No surgical complications, such as skin necrosis or traumatic extrusion, have yet been reported in the 6 patients who underwent this procedure. Eliminating the softband reduces complaints of pressure and those of tension headaches, decreases the frequency of pressure ulcers associated with the device, and improves aesthetic appeal and overall user compliance [29,30]. An implanted Ponto should thus replace the softband when the cortical bone of the skull has sufficiently thickened for osseointegration of the titanium implant. In this study, the implanted Ponto led to an improvement of 15 ± 4.72 dB in the hearing threshold when compared to the softband Ponto. This was mostly the result of less energy loss, as sound is directly transmitted through the screw. Since children have a more critical need for hearing during their developmental years than adults in order to understand everyday speech [31], the improvement in hearing achieved using the implanted Ponto when compared to the Ponto softband is essential for language development in children. We used the Mandarin testing system to evaluate the speech discrimination score under the softband and implanted Ponto conditions. The implanted Ponto led to better speech discrimination ability. This suggests that the implanted Ponto leads to better hearing performance than the Ponto softband. In order to improve hearing, patients could undergo Ponto implantation as soon as the thickness of the cortical bone is sufficient for implantation.
References [1] K. Deng, L. Dai, L. Yi, C. Deng, X. Li, J. Zhu, Birth defects research. Part A, Clin. Mol. Teratol. 2016 (106) (2016) 88–94. [2] H.F. Schuknecht, Laryngoscope 1989 (99) (1989) 908–917. [3] W.C. Hall, Matern. Child Health J. 2017 (21) (2017) 961–965. [4] D. Li, W. Chin, J. Wu, Q. Zhang, F. Xu, Z. Xu, R. Zhang, Aesthetic Plast. Surg. 2010 (34) (2010) 570–576. [5] A.J. Bosman, A.F. Snik, M.K. Hol, E.A. Mylanus, J. Am. Acad. Audiol. 2013 (24) (2013) 505–513. [6] D.H. Coelho, A.J. Tampio, Ann. Otol. Rhinol. Laryngol. 2017 (126) (2017) [61]–[66]. [7] T.G. Calon, M. van Hoof, H. van den Berge, A.J. de Bruijn, J. van Tongeren, J.R. Hof, J.W. Brunings, S. Jonhede, L.J. Anteunis, M. Janssen, M.A. Joore, M. Holmberg, M.L. Johansson, R.J. Stokroos, Trials 2016 (17) (2016) 540. [8] Y. Fan, Y. Zhang, P. Wang, Z. Wang, X. Zhu, H. Yang, X. Chen, JAMA Otolaryngol. Head Neck Surg. 2014 (140) (2014) 357–362. [9] Y. Wang, X. Chen, Y. Fan, Z. Wang, Lin chuang er bi yan hou tou jing wai ke za zhi, J. Clin. Otorhinolaryngol. Head Neck Surg. 2015 (29) (2015) 291–294. [10] H. Weerda, Facial Plast. Surg.: FPS 1988 (5) (1988) 385–388. [11] Y. Zheng, S.D. Soli, K. Wang, J. Meng, Z. Meng, K. Xu, Y. Tao, Audiol. Neuro. Otol. 2009 (14) (2009) 214–222. [12] W. Wu, H. Zhang, J. Chen, J. Chen, C. Lin, Comput. Biol. Med. 2011 (41) (2011) 131–138. [13] N. Skotara, U. Salden, M. Kugow, B. Hanel-Faulhaber, B. Roder, BMC Neurosci. 2012 (13) (2012) 44. [14] B.W. Kesser, K. Krook, L.C. Gray, Laryngoscope 2013 (123) (2013) 2270–2275. [15] R. Pujol, M. Lavigne-Rebillard, Int. J. Pediatr. Otorhinolaryngol. 1995 32 (Suppl) (1995) S177–S182. [16] Y. Zhong, T. Xu, R. Dong, J. Lyu, B. Liu, X. Chen, Int. J. Pediatr. Otorhinolaryngol. 2017 (96) (2017) 106–110. [17] E. Vyskocil, R. Liepins, A. Kaider, M. Blineder, S. Hamzavi, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2017 (38) (2017) 318–324. [18] B.W. Kesser, E.D. Cole, L.C. Gray, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2016 (37) (2016) 499–503. [19] H. Byun, I.J. Moon, S.Y. Woo, S.H. Jin, H. Park, W.H. Chung, S.H. Hong, Y.S. Cho, Ear Hear. 2015 (36) (2015) e183–189. [20] R.C. Nelissen, E.A. Mylanus, C.W. Cremers, M.K. Hol, A.F. Snik, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2015 (36) (2015) 826–833. [21] I.J. Moon, H. Byun, S.H. Jin, S. Kwon, W.H. Chung, S.H. Hong, Y.S. Cho, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2014 (35) (2014) 639–644. [22] M.K. Hol, A.F. Snik, E.A. Mylanus, C.W. Cremers, Audiol. Neuro. Otol. 2005 (10) (2005) 159–168. [23] A. van Wieringen, K. De Voecht, A.J. Bosman, J. Wouters, Clin. Otolaryngol.: Official J. ENT-UK ; Offic. J. Neth. Soc. Oto-Rhino-Laryngol. Cervico-Facial Surg. 2011 (36) (2011) 114–120. [24] E.R. Oliver, B.B. Hughley, D.C. Shonka, B.W. Kesser, Otology & neurotology : official publication of the American otological society, Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2011 (32) (2011) 252–258. [25] R.A. Jahrsdoerfer, P.R. Lambert, Am. J. Otol. 1998 (19) (1998) 283–287. [26] Y. Fan, Y. Zhang, S. Wang, X. Chen, Int. J. Pediatr. Otorhinolaryngol. 2014 (78) (2014) 60–64. [27] S.E. Han, S.Y. Lim, J.K. Pyon, S.I. Bang, G.H. Mun, K.S. Oh, J. Plast., Reconstr. Aesthetic Surg. JPRAS 2015 (68) (2015) 1085–1094. [28] A. Steffen, S. Klaiber, R. Katzbach, S. Nitsch, I.R. Konig, H. Frenzel, Aesthetic Surg. J. 2008 (28) (2008) 404–411. [29] J.B. Spitzer, S.N. Ghossaini, J.J. Wazen, Am. J. Audiol. 2002 (11) (2002) 96–103. [30] S. Lloyd, J. Almeyda, K.S. Sirimanna, D.M. Albert, C.M. Bailey, J. Laryngol. Otol. 2007 (121) (2007) 826–831. [31] C. Fuchsmann, S. Tringali, F. Disant, G. Buiret, C. Dubreuil, P. Froehlich, E. Truy, Acta oto-Laryngol. 2010 (130) (2010) 1343–1351.
5. Conclusion Using softband bone-anchored hearing devices is effective for the improvement of the audition in infants with bilateral microtia and aural atresia. Wearing the softband bone-anchored hearing device before auricle reconstruction and combining bone-anchored hearing device implantation with auricle reconstructive surgery may be the optimal clinical choice for children with bilateral microtia and aural atresia. This approach may lead to significant hearing improvement and minimal surgical and anesthetic injuries. Funding This work was supported by a grant to Xiaowei Chen from the General Programs of National Natural Science Foundation of China (grant number: 81271053) and the Emergency Management Programs of the National Natural Science Foundation of China (grant number: 81450026). Conflicts of interest The authors report no conflicts of interest and have received no payment for the preparation of this manuscript. The authors alone are responsible for the content and writing of this paper. Authors' contributions XWC conceived and designed the experiments. YBW and XMF
125
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Hearing improvement with softband and implanted bone-anchored hearing devices and modified implantation surgery in patients with bilateral microtia-atresia
T
Yibei Wang, Xinmiao Fan, Pu Wang, Yue Fan, Xiaowei Chen∗ Department of Otolaryngology, Peking Union Medical College Hospital, Beijing, 100730, China
A R T I C L E I N F O
A B S T R A C T
Keywords: Bilateral microtia-atresia Bone anchored hearing devices Auditory development Modified implantation surgery Hearing improvement Speech discrimination
Objective: To evaluate auditory development and hearing improvement in patients with bilateral microtiaatresia using softband and implanted bone-anchored hearing devices and to modify the implantation surgery. Methods: The subjects were divided into two groups: the softband group (40 infants, 3 months to 2 years old, Ponto softband) and the implanted group (6 patients, 6–28 years old, Ponto). The Infant-Toddler Meaning Auditory Integration Scale was used conducted to evaluate auditory development at baseline and after 3, 6, 12, and 24 months, and visual reinforcement audiometry was used to assess the auditory threshold in the softband group. In the implanted group, bone-anchored hearing devices were implanted combined with the auricular reconstruction surgery, and high-resolution CT was used to assess the deformity preoperatively. Auditory threshold and speech discrimination scores of the patients with implants were measured under the unaided, softband, and implanted conditions. Results: Total Infant-Toddler Meaning Auditory Integration Scale scores in the softband group improved significantly and approached normal levels. The average visual reinforcement audiometry values under the unaided and softband conditions were 76.75 ± 6.05 dB HL and 32.25 ± 6.20 dB HL (P < 0.01), respectively. In the implanted group, the auditory thresholds under the unaided, softband, and implanted conditions were 59.17 ± 3.76 dB HL, 32.5 ± 2.74 dB HL, and 17.5 ± 5.24 dB HL (P < 0.01), respectively. The respective speech discrimination scores were 23.33 ± 14.72%, 77.17 ± 6.46%, and 96.50 ± 2.66% (P < 0.01). Conclusions: Using softband bone-anchored hearing devices is effective for auditory development and hearing improvement in infants with bilateral microtia-atresia. Wearing softband bone-anchored hearing devices before auricle reconstruction and combining bone-anchored hearing device implantation with auricular reconstruction surgery may bethe optimal clinical choice for these patients, and results in more significant hearing improvement and minimal surgical and anesthetic injury.
1. Introduction Congenital microtia-atresia results from developmental defects of the auricle and is generally associated with malformations of the external auditory canal and the middle ear. The incidence of congenital microtia-atresia is estimated to be between 0.81 in 10,000 and 3.06 in 20,000 live births in China [1]. Approximately 10% of patients with congenital microtia-atresia have the condition in both ears. Patients with congenital microtia-atresia often experience conductive hearing loss with an air-bone gap of 50–60 dB due to bony or soft-tissue occlusion [2]. The first two years of life are critical in language acquisition. During
this period, hearing ability is crucial in learning spoken language. There is growing evidence that the absence of sound and language access leads to cognitive delay, mental health difficulties, a higher possibility of traumatic accidents, and limited literacy [3]. Early intervention for children with hearing impairment is thus essential. Patients with microtia-atresia have lower self-esteem and social dysfunction [4], and require hearing rehabilitation and auricular reconstruction surgery to improve their appearance. Until now, the use of bone-anchored hearing devices has been one of the most reliable methods for auditory rehabilitation [5,6]. Softband bone-anchored hearing devices fix the processor around the head via a soft band. Sound is thus transmitted directly by the skull bone to the
∗
Corresponding author. Department of Otolaryngology, Peking Union Medical College Hospital, #1 shuaifuyuan, Beijing, 100730, China. E-mail addresses: [email protected] (Y. Wang), [email protected] (X. Fan), [email protected] (P. Wang), [email protected] (Y. Fan), [email protected] (X. Chen). https://doi.org/10.1016/j.ijporl.2017.11.010 Received 27 September 2017; Received in revised form 30 October 2017; Accepted 1 November 2017 Available online 14 November 2017 0165-5876/ © 2017 Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
includes 10 items grouped into 3 domains: vocalization behavior (items 1 and 2), alerting to sounds (items 3–6), and deriving meaning from sound (items 7–10). Each question has five different response alternatives: 0 = never, 1 = rarely, 2 = occasionally, 3 = frequently, and 4 = always [11]. The Infant-Toddler Meaningful Auditory Integration Scale was used to evaluate auditory development at baseline (0 months), and 6, 12, and 24 months after wearing the Ponto softband. Visual reinforcement audiometry was used to assess the auditory threshold after wearing the Ponto softband for 6 months under unaided and aided conditions.
inner ear. Implanted bone-anchored hearing devices transmit sound using a titanium implant, which couples the vibration transducer to the skull bone via a skin-penetrating abutment [7]. Operation timing and the position of implanted bone-anchored hearing devices are crucial. Our group has previously studied hearing improvement following the use of softband and implanted bone-anchored hearing devices in patients with bilateral microtia-atresia [8]. We have also recommended the early use of softband bone-anchored hearing devices in infants with bilateral microtia-atresia [9]. The growth curve describing auditory development in infants with bilateral microtia-atresia using softband bone-anchored hearing devices is yet unknown. Choosing the operation time and implantation position are also major challenges for surgeons. Here we aimed to evaluate the auditory development of Mandarinspeaking children with bilateral microtia-atresia using the Ponto softband by following up 40 infants over two years. We also aimed to modify the implantation surgery by combining it with auricle reconstruction.
2.2.2. Implanted group 2.2.2.1. Imaging evaluation and modified implantation surgery. All patients underwent preoperative temporal bone high-resolution computed tomography to evaluate temporal bone thickness and the middle ear structures. Patients were graded using the Jahrsdoerfer grading scale. According to Jahrsdoerfer criteria, 6 patients who scored less than 6 points in both ears were considered poor candidates for hearing reconstruction. Therefore, atresiaplasty was not performed in these patients. In the first stage of auricular reconstruction surgery, a soft-tissue skin expander was implanted in the mastoid region to enable us to obtain a sufficient amount of skin. In the second stage, an autogenous rib cartilage framework was carved to reconstruct the auricle. The third stage comprised the second revision of the auricular reconstruction. Ponto implantation was performed during the auricular reconstructive surgery, especially the second stage. In patients who had undergone the second stage of surgery, we were able to combine Ponto implantation with the third stage. In patients who required 2-stage implantation, the titanium implant was inserted into the mastoid bone at the end of the soft-tissue skin expander or the auricle reconstruction sugeries. The abutment was attached to the titanium screw during the next stage of auricle reconstructions, which was performed 4–6 months later.
2. Methods 2.1. Subjects This single center retrospective study included 46 patients with bilateral microtia-atresia who were enrolled at Peking Union Medical College Hospital in Beijing, China, from January 1, 2014, to April 30, 2016. The study subjects comprised 40 infants (aged 3 months to 2 years) and 6 patients with implants (aged 6–28 years). Ethical approval for this study was obtained from the Institutional Review Board of Peking Union Medical College Hospital. Parents of all patients in the study provided written informed consent for the use of soft-band and implanted bone-anchored hearing devices and the tests. 2.1.1. Softband group The softband group consisted of 40 infants with bilateral microtiaatresia comprising 23 boys and 17 girls ranging in age from 3 months to 2 years (average age, 8.45 ± 5.67 months; median age, 7 months), wearing Ponto softband (Ponto pro, made by Oticon). All 40 patients were classified as grade III according to the Marx classification of the severity of deformity and bilateral aural atresia [10]. The average bone conduction threshold of the deformed ear, as assessed using the auditory brainstem response, was 18.13 ± 4.84 dB nHL (range, 0–25 dB nHL) and the average air conduction threshold was 76.38 ± 4.53 dB nHL (range, 70–85 dB nHL).
2.2.2.2. Audiological evaluation. Sound field pure-tone audiometry was conducted for all patients at 250, 500, 1,000, 2,000, 4,000, and 8000 Hz under unaided, softband-aided, and implantation-aided conditions. The speech perception tests were conducted in a sound field using the Mandarin speech test system [12]. A bisyllabic test list was used to obtain speech discrimination scores under the unaided, softband, and Ponto-implanted conditions. All test materials were presented twice. The average score was used in the final analysis. 2.3. Statistical analysis
2.1.2. Implanted group Six patients aged 6–28 years (average age, 15.17 ± 10.00 years) received Ponto (Ponto, made by Oticon) implantation surgery between May 2015 and December 2016 after using the Ponto softband (Ponto pro, made by Oticon) for 3–12 months (mean, 6 months). All 6 patients, who were male, were classified as grade III according to the Marx classification of the severity of deformity and bilateral aural atresia [10]. The average bone conduction threshold of the deformed ear, as assessed using pure tone audiometry, was 17.50 ± 2.74 dB HL (range, 0–20 dB HL), and the average air conduction threshold was 76.67 ± 5.16 dB HL (range, 70–85 dB HL) for tones between 250 and 8000 Hz. One patient had undergone auricle reconstruction surgery in 1992 in another hospital. This patient underwent bilateral bone-anchored hearing device implantation.
All data were analyzed using SPSS (version 19.0, IBM Corp.). Continuous variables are presented as mean ± SD. Infant-Toddler Meaningful Auditory Integration Scale scores were compared to normal scores for this scale [11]. Repeated measures ANOVAs with Greenhouse-Geisser corrections were used to assess differences between the standard and softband-aided Infant-Toddler Meaningful Auditory Integration Scale scores. Paired t tests were used to assess the differences between patients with and without Ponto softbands in the softband group. ANOVAs were used to assess differences among the unaided, softband-aided, and implant-aided conditions in the implanted group. P values < 0.01 were considered statistically significant. 3. Results
2.2. Main outcome measures
3.1. Auditory development in infants using Ponto softbands
2.2.1. Softband group Auditory and speech development of infants were mainly assessed using the translated Chinese version of the Infant-Toddler Meaningful Auditory Integration Scale. The Infant-Toddler Meaningful Auditory Integration Scale is a structured parent interview questionnaire that
The Infant-Toddler Meaningful Auditory Integration Scale scores of the 40 children in the softband group were assessed at baseline (0 month), and 3, 6, 12, and 24 months after baseline. The mean follow-up time was 21.9 ± 4.6 months. 17.5% of the patients were lost to followup. The patients wore the devices for an average of 6.57 ± 1.22 h per 121
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
Fig. 1. IT-MAIS Total Score In this figure, the upper red curve refers to the scores of normal level, while the broken lines referringreferring to the scores of 40 microtia-atresia infants. ITMAIS total scores of these infants were lower than the standard, which were found to have improved significantly and approaching the normal level gradually with time after wearing the softband Ponto.For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.
3.3. Hearing improvement
day. As shown in Fig. 1, Infant-Toddler Meaningful Auditory Integration Scale total scores in the softband group were lower than the standard scores [11], although they improved significantly and approached normal levels gradually with time. The average difference in InfantToddler Meaningful Auditory Integration Scale scores of the patients in the softband group and standard scores at baseline (0 months), and 3, 6, 12, and 24 months after baseline were 44.30 ± 15.31%, 38.15 ± 12.87%, 29.18 ± 10.95%, 19.80 ± 10.55% and 5.24 ± 11.55%, respectively. The difference was greatest at baseline and decreased gradually with time. This change was statistically significant (Greenhouse-Geisser, F = 144.09, P < 0.001) (Fig. 2).
3.3.1. Hearing threshold In the softband group, the average unaided hearing thresholds measured using visual reinforcement audiometry were 76.75 ± 6.05 dB HL, while the average hearing thresholds when using a Ponto softband were 32.26 ± 6.20 dB HL. There was a statistically significant improvement of 44.50 ± 3.89 dB HL in the threshold, as assessed using a paired t-test (t = 72.36, P < 0.01). In the implanted group, the mean preoperative sound field threshold was 59.17 ± 3.76 dB HL in patients with unaided hearing. The mean softband and postoperative sound field thresholds were 32.5 ± 2.74 dB HL, and 17.5 ± 5.24, respectively. These values were significantly different (F = 163.05, P < 0.001). The Ponto softband and the implantation procedure resulted in mean hearing improvements of 26.67 ± 4.08 dB and 41.67 ± 5.16 dB. The implanted Ponto resulted in an improvement greater than that achieved using the softband by 15 ± 4.72 dB, as shown in http://jamanetwork.com/ journals/jamaotolaryngology/fullarticle/1816004 Fig. 5.
3.2. Modified implantation surgery The average Jahrsdoerfer grading scale score was 4.5 ± 0.55. All patients had stapes abnormalities. Of the 6 patients in the group, 5 had undergone Ponto implantation combined with auricular reconstructive surgery. One patient who already had reconstructed auricles underwent bilateral implantation in one surgery using the conventional surgical method. Among the patients in this group, 2 children younger than 7 years of age required 2-stage surgeries for implantation. In one of these children, we inserted the titanium implant at the end of the soft-tissue skin expander surgery and attached the abutment at the end of the auricular reconstruction surgery (Fig. 3). In the other child, we inserted the titanium implant at the end of the second stage of the auricular reconstruction surgery and attached the abutment at the end of the third stage of the surgery. For the other 3 patients, we inserted the titanium implant and attach it to the abutment at the end of the second stage of the auricular reconstruction surgery (Fig. 4). At this time, no skin necrosis and or traumatic extrusions have been reported.
3.3.2. Speech discrimination score In the implanted group, the mean unaided speech discrimination score, as evaluated using the bisyllabic test (presentation level, 65 dB HL) was 23.33 ± 14.72%. The mean score was 77.17 ± 6.46% in patients using the Ponto softband and 96.50 ± 2.66%in patients with implanted Ponto devices. These improvements of 53.84 ± 15.29% and 73.17 ± 15.29% were statistically significant (F = 97.45, P < 0.001). Ponto implantation led to a greater improvement 19.33 ± 4.89% than the softband, as shown in http://jamanetwork. com/journals/jamaotolaryngology/fullarticle/1816004 Fig. 6. 122
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
Fig. 2. The difference of the IT-MAIS Scores Fig. 2 showed the differences of the IT-MAIS scores compared to the standard scores after wearing the softband Ponto for baseline (0 months), three months, six months, twelve months and twenty-four months. The difference was greatest in baseline and decrease gradually with time, which is statistically significant (Greenhouse-Geisser, F = 144.09,P < 0.001).
4. Discussion
bone-anchored hearing devices in a two-year follow-up study of 40 infants. We found that the softband bone-anchored hearing device had effective impacts on auditory development. Using questionnaires completed by the patient's parents is the best way to assess early prelingual auditory development and the efficacy of hearing intervention, as younger children have difficulty performing other types of tests used to assess auditory development. The InfantToddler Meaningful Auditory Integration Scale has been reported to have good reliability and validity in measuring auditory development and hearing rehabilitation efficacy [16]. A study of 120 infants and toddlers with normal hearing born to Mandarin-speaking parents was used to establish normal scores for the spontaneous detection, recognition, and discrimination of sounds. This study provides us with a trajectory for standard Infant-Toddler Meaningful Auditory Integration Scale total scores during the first two years of life [11]. We found that infants with bilateral microtia-atresia had deficits in auditory development, with a median decrease of 44.30 ± 15.31% in the Infant-Toddler Meaningful Auditory Integration Scale total score. This finding reflects the necessity for early intervention to improve hearing in infants with bilateral microtia-atresia. The current treatment options for bilateral microtia-atresia are varied but limited. They include surgical reconstruction of the external auditory canal and boneanchored hearing device implantation. Bone-anchored hearing device
4.1. The efficacy and superiority of softband bone-anchored hearing devices in children with bilateral microtia-atresia Hearing plays a crucial role in the early stages of language development. Inadequate access to spoken language may delay the development of neuro-linguistic structures of the brain, and especially grammar and second language acquisition [13]. The severe conductive hearing loss associated with bilateral microtia-atresia gives rise to communication barriers and delays in speech-language abilities. Recent evidence suggests that children with microtia-atresia are at risk for delays in speech-language development and cognition, and behavioral problems that can affect functioning in school [14]. Additionally, development of the central auditory system might be altered due to the limited stimulation caused by the significant air-bone gap in these patients [15]. There is consensus that early hearing rehabilitation contributes to auditory and language development. To our knowledge, there are yet no studies incorporating a large sample and follow-up observation to investigate auditory development in patients with bilateral microtia-atresia using the Ponto softband bone-anchored hearing device. We aimed to evaluate auditory development in Mandarin-speaking patients with bilateral microtia-atresia using softband
Fig. 3. The modified 2-stage Ponto implantation surgery Fig. 3 showed the child who was inserted the titanium implant at the end of the soft-tissue skin expander surgery and attached the abutment at the end of the second stage of the auricular reconstruction surgery. A and B showed the titanium implant which was inserted in the soft –tissue skin expender surgery. C showed the attachment of the abutment.
123
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
Fig. 4. The modified implantation surgery Fig. 4 showed the Ponto implantation surgery combined with the second stage of the auricular reconstruction surgery. We inserted the titanium implant and attach it to the abutment after the auricle was settled, as shown in A. The skin above the abutment was eliminated by skin punch to make the minimal injury as shown in B. The healing cap was used to reduce the infection as shown in C.
Fig. 5. Hearing Thresholds of the Implanted Group Fig. 5 showed the mean hearing thresholds in the implanted group under the unaided, softband and implanted conditions. The soft-band Ponto and the implantation gave a mean hearing improvement of 26.67 ± 4.08 dB and 41.67 ± 5.16 dB, which was statistically significant (P < 0.001).
Fig. 6. The Speech Discrimination Score of the Implanted Group Fig. 6 showed the mean speech discrimination in the implanted group under the unaided, softband and implanted conditions. The softband and the implantation gave a mean hearing improvement of 53.84 ± 15.29% and 73.17 ± 15.29%, which was statistically significant (P < 0.001).
implantation and aural reconstruction surgeries certainly improve speech recognition ability [17–23]. It is undeniable that there are many postoperative complications following aural reconstruction surgery, such as facial nerve injury and meatal stenosis [24,25]. In patients with microtia-atresia, aural reconstruction surgery may lead to deficits in the blood supply of the flap used in the secondary stage of auricle reconstruction surgery. This may in turn increase surgical difficulties and complications postoperatively. The use of softband bone-anchored hearing devices is a common clinical choice for the treatment of infants with bilateral microtiaatresia. These devices are used as early as 3 months of age, before the patient's skull bones are sufficiently thick for implantation surgery. After wearing the softband bone-anchored hearing device for 6 months, most patients experienced improved auditory development, and the developmental trajectories of the patients gradually neared or reached those of healthy infants and children. This finding is comparable to that of our previous study [26]. In the current study, we further characterized the auditory developmental trajectories of infants with bilateral microtia-atresia wearing softband bone-anchored hearing devices. Auditory development is encouraging in these patients. The hearing threshold was 32.26 ± 6.20 dBHL, as tested using visual reinforcement audiometry, which is an improvement of 44.50 ± 3.89 dB.
Considering the demand for hearing improvement and the surgical complications described above, we recommend the use of softband bone-anchored hearing devices to improve hearing ability before auricular reconstruction surgery becomes the optimal treatment choice in children with bilateral microtia-atresia.
4.2. Modified surgery combining bone-anchored hearing device implantation with auricle reconstruction Auricular reconstruction surgery using autologous cartilage is currently the most effective method used to correct deformities due to microtia [27]. This procedure results in both aesthetic and psychosocial improvements. As discussed above, hearing rehabilitation is essential in patients with bilateral microtia-atresia [28]. Ponto implantation was performed at the end of the surgery after the auricle was settled. The position of the screw should be carefully determined to prevent the sound processor from impeding the auricular reconstruction and impairing the blood supply of the flap. We always settle the screw 5–7 cm away from the tragus of the reconstructed auricle at a 45-degree angle. We do not remove subcutaneous tissue during this procedure. We use a punch to remove the skin above the screw in order to establish contact with the abutment. Combining Ponto implantation with the second 124
International Journal of Pediatric Otorhinolaryngology 104 (2018) 120–125
Y. Wang et al.
administered the IT-MAIS questionnaire and collected the data. YBW and PW tested the hearing thresholds and assigned speech discrimination scores. YBW analyzed the data. YBW wrote the paper. XWC and YF revised the paper critically for important intellectual content. All authors read and approved the final manuscript.
stage of ear reconstruction helps position the screw and can spare the patients from additional incision and anesthetic use. Some young children require 2-stage implantation because the thinner and less dense skull bone requires a period of 4–6 months for osseointegration of the fixture. In this study, 2 patient younger than 7 years of age underwent 2-stage implantation. Combining the 2 stages of the implantation with 2 steps of the auricular reconstruction surgery reduced the total time of treatment. No surgical complications, such as skin necrosis or traumatic extrusion, have yet been reported in the 6 patients who underwent this procedure. Eliminating the softband reduces complaints of pressure and those of tension headaches, decreases the frequency of pressure ulcers associated with the device, and improves aesthetic appeal and overall user compliance [29,30]. An implanted Ponto should thus replace the softband when the cortical bone of the skull has sufficiently thickened for osseointegration of the titanium implant. In this study, the implanted Ponto led to an improvement of 15 ± 4.72 dB in the hearing threshold when compared to the softband Ponto. This was mostly the result of less energy loss, as sound is directly transmitted through the screw. Since children have a more critical need for hearing during their developmental years than adults in order to understand everyday speech [31], the improvement in hearing achieved using the implanted Ponto when compared to the Ponto softband is essential for language development in children. We used the Mandarin testing system to evaluate the speech discrimination score under the softband and implanted Ponto conditions. The implanted Ponto led to better speech discrimination ability. This suggests that the implanted Ponto leads to better hearing performance than the Ponto softband. In order to improve hearing, patients could undergo Ponto implantation as soon as the thickness of the cortical bone is sufficient for implantation.
References [1] K. Deng, L. Dai, L. Yi, C. Deng, X. Li, J. Zhu, Birth defects research. Part A, Clin. Mol. Teratol. 2016 (106) (2016) 88–94. [2] H.F. Schuknecht, Laryngoscope 1989 (99) (1989) 908–917. [3] W.C. Hall, Matern. Child Health J. 2017 (21) (2017) 961–965. [4] D. Li, W. Chin, J. Wu, Q. Zhang, F. Xu, Z. Xu, R. Zhang, Aesthetic Plast. Surg. 2010 (34) (2010) 570–576. [5] A.J. Bosman, A.F. Snik, M.K. Hol, E.A. Mylanus, J. Am. Acad. Audiol. 2013 (24) (2013) 505–513. [6] D.H. Coelho, A.J. Tampio, Ann. Otol. Rhinol. Laryngol. 2017 (126) (2017) [61]–[66]. [7] T.G. Calon, M. van Hoof, H. van den Berge, A.J. de Bruijn, J. van Tongeren, J.R. Hof, J.W. Brunings, S. Jonhede, L.J. Anteunis, M. Janssen, M.A. Joore, M. Holmberg, M.L. Johansson, R.J. Stokroos, Trials 2016 (17) (2016) 540. [8] Y. Fan, Y. Zhang, P. Wang, Z. Wang, X. Zhu, H. Yang, X. Chen, JAMA Otolaryngol. Head Neck Surg. 2014 (140) (2014) 357–362. [9] Y. Wang, X. Chen, Y. Fan, Z. Wang, Lin chuang er bi yan hou tou jing wai ke za zhi, J. Clin. Otorhinolaryngol. Head Neck Surg. 2015 (29) (2015) 291–294. [10] H. Weerda, Facial Plast. Surg.: FPS 1988 (5) (1988) 385–388. [11] Y. Zheng, S.D. Soli, K. Wang, J. Meng, Z. Meng, K. Xu, Y. Tao, Audiol. Neuro. Otol. 2009 (14) (2009) 214–222. [12] W. Wu, H. Zhang, J. Chen, J. Chen, C. Lin, Comput. Biol. Med. 2011 (41) (2011) 131–138. [13] N. Skotara, U. Salden, M. Kugow, B. Hanel-Faulhaber, B. Roder, BMC Neurosci. 2012 (13) (2012) 44. [14] B.W. Kesser, K. Krook, L.C. Gray, Laryngoscope 2013 (123) (2013) 2270–2275. [15] R. Pujol, M. Lavigne-Rebillard, Int. J. Pediatr. Otorhinolaryngol. 1995 32 (Suppl) (1995) S177–S182. [16] Y. Zhong, T. Xu, R. Dong, J. Lyu, B. Liu, X. Chen, Int. J. Pediatr. Otorhinolaryngol. 2017 (96) (2017) 106–110. [17] E. Vyskocil, R. Liepins, A. Kaider, M. Blineder, S. Hamzavi, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2017 (38) (2017) 318–324. [18] B.W. Kesser, E.D. Cole, L.C. Gray, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2016 (37) (2016) 499–503. [19] H. Byun, I.J. Moon, S.Y. Woo, S.H. Jin, H. Park, W.H. Chung, S.H. Hong, Y.S. Cho, Ear Hear. 2015 (36) (2015) e183–189. [20] R.C. Nelissen, E.A. Mylanus, C.W. Cremers, M.K. Hol, A.F. Snik, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2015 (36) (2015) 826–833. [21] I.J. Moon, H. Byun, S.H. Jin, S. Kwon, W.H. Chung, S.H. Hong, Y.S. Cho, Otolo. Neurotol.: Official Publ. Am. Otol. Soc. Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2014 (35) (2014) 639–644. [22] M.K. Hol, A.F. Snik, E.A. Mylanus, C.W. Cremers, Audiol. Neuro. Otol. 2005 (10) (2005) 159–168. [23] A. van Wieringen, K. De Voecht, A.J. Bosman, J. Wouters, Clin. Otolaryngol.: Official J. ENT-UK ; Offic. J. Neth. Soc. Oto-Rhino-Laryngol. Cervico-Facial Surg. 2011 (36) (2011) 114–120. [24] E.R. Oliver, B.B. Hughley, D.C. Shonka, B.W. Kesser, Otology & neurotology : official publication of the American otological society, Am. Neurotol. Soc. Eur. Acad. Otol. Neurotol. 2011 (32) (2011) 252–258. [25] R.A. Jahrsdoerfer, P.R. Lambert, Am. J. Otol. 1998 (19) (1998) 283–287. [26] Y. Fan, Y. Zhang, S. Wang, X. Chen, Int. J. Pediatr. Otorhinolaryngol. 2014 (78) (2014) 60–64. [27] S.E. Han, S.Y. Lim, J.K. Pyon, S.I. Bang, G.H. Mun, K.S. Oh, J. Plast., Reconstr. Aesthetic Surg. JPRAS 2015 (68) (2015) 1085–1094. [28] A. Steffen, S. Klaiber, R. Katzbach, S. Nitsch, I.R. Konig, H. Frenzel, Aesthetic Surg. J. 2008 (28) (2008) 404–411. [29] J.B. Spitzer, S.N. Ghossaini, J.J. Wazen, Am. J. Audiol. 2002 (11) (2002) 96–103. [30] S. Lloyd, J. Almeyda, K.S. Sirimanna, D.M. Albert, C.M. Bailey, J. Laryngol. Otol. 2007 (121) (2007) 826–831. [31] C. Fuchsmann, S. Tringali, F. Disant, G. Buiret, C. Dubreuil, P. Froehlich, E. Truy, Acta oto-Laryngol. 2010 (130) (2010) 1343–1351.
5. Conclusion Using softband bone-anchored hearing devices is effective for the improvement of the audition in infants with bilateral microtia and aural atresia. Wearing the softband bone-anchored hearing device before auricle reconstruction and combining bone-anchored hearing device implantation with auricle reconstructive surgery may be the optimal clinical choice for children with bilateral microtia and aural atresia. This approach may lead to significant hearing improvement and minimal surgical and anesthetic injuries. Funding This work was supported by a grant to Xiaowei Chen from the General Programs of National Natural Science Foundation of China (grant number: 81271053) and the Emergency Management Programs of the National Natural Science Foundation of China (grant number: 81450026). Conflicts of interest The authors report no conflicts of interest and have received no payment for the preparation of this manuscript. The authors alone are responsible for the content and writing of this paper. Authors' contributions XWC conceived and designed the experiments. YBW and XMF
125