Otology & Neurotology

Speech Perception Growth Patterns in Prelingual Deaf Children With Bilateral Sequential Cochlear Implantation Objective: To evaluate speech perception following the first (CI-1) and second (CI-2) cochlear implantation (CI) in children with sequential bilateral CI. Study Design: Retrospective. Patients: Seventy children with follow-up for 60 months post CI-1 and 36 months post CI-2. Main Outcome Measures: Word recognition score (WRS) was the main outcome. WRSs were compared by age at CI operation (group A ≤ 3.5 yr, B 3.6–8.6, for CI-1; group I ≤ 3.5 yr, II 3.6–7.0, III 7.1–13, IV > 13, for CI-2). Results: For CI-1, the WRS of group A exceeded 80% at 24 months post procedure, earlier than group B (54 mo). Group A also had a shorter period of CI-1 use up to the WRS plateau than group B. CI-2 showed an initial burst of WRS growth much earlier than CI-1. This initial burst was most robust within 3 months in group II, but modest in group IV. The periods of CI-2 use (11–17 mo) up to the WRS plateau were much shorter than CI-1 (40–64 mo). Group I did not show the best WRS at 1 month post CI but later exceeded the other groups. Conclusion: Children received an immediate benefit by a burst of WRS growth from CI-2 earlier than CI-1, even within 3 months, suggesting that CI-1 gets the auditory cortex ready to foster speech processing from CI-2. The CI-2 performance depends on age at CI-2 implantation and on CI-1 performance. Our current findings will be relevant for clinicians who are counselling parents on CI-2 surgery. Address correspondence and reprint requests to Hong Ju Park, M.D., Ph.D., Department of Otorhinolaryngology-Head and Neck Surgery, Asan Medical Center, University of Ulsan College of Medicine, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Republic of Korea; E-mail: dzness@hotmail.com The authors disclose no conflicts of interest. Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Long-term outcomes of bone conduction hearing implants in patients with bilateral microtia-atresia Objectives: To evaluate the long-term outcomes of three different types of bone conduction hearing implants (BCHI)—BAHA, Ponto, and Bonebridge—in Mandarin-speaking patients with bilateral microtia-atresia. Methods: This cohort study enrolled 59 patients affected by bilateral microtia-atresia, with an upper bone conduction threshold limit of 30 dB HL at frequencies of 0.5 to 4 kHz. All subjects underwent unilateral BCHI surgery, including 26 (18 males, 8 females, of mean age 8.7 ± 1.9 yr) implanted with BAHA devices; 10 (7 males, 3 females, of mean age 11.7 ± 2.8 yr) implanted with Ponto devices; and 23 (14 males, 9 females, of mean age 9.0 ± 1.8 yr) implanted with Bonebridge devices. The main outcome measures included long-term audiological benefits, patient satisfaction, and complications. Each subject acted as his or her own control. Results: Two years after BCHI surgery, the mean hearing thresholds in the BAHA, Ponto, and Bonebridge groups had improved to 22.6 ± 1.6 dB HL, 21.6 ± 1.2 dB HL, and 22.5 ± 1.5 dB HL, respectively. The mean percentages of subjects in these three groups recognizing speech at 65 dB SPL under quiet conditions were 97.7 ± 4.2%, 96.3 ± 1.1%, and 94.4 ± 9.4%, respectively, whereas the mean percentages recognizing speech under noise conditions (signal:noise ratio +5) were 87.0 ± 1.8%, 89.3 ± 9.3%, and 85.3 ± 4.7%, respectively. Questionnaires revealed patients' benefits and satisfaction with this surgery. Three (11.5%) of 26 patients in the BAHA group and 1 (10%) of 10 in the Ponto group experienced skin irritation, but all recovered after local treatment. Five (19.2%) patients in the BAHA group and two (20%) in the Ponto experienced abutment extrusion about 6 months postoperatively, with all achieving good results after revision surgery to replace the abutment. One (3.8%) patient in the BAHA group experienced local chronic inflammation and underwent surgery to replace the BAHA with a Bonebridge implant. One (4.3%) patient in the Bonebridge group developed a local infection 3 months postoperatively and underwent implant removal. Conclusions: All three BCHIs were well tolerated after long-term follow-up, and all improved audiometric thresholds and the intelligibility of speech in the presence of both quiet and noise. These implants should be considered valid and safe options for the functional rehabilitation of patients with bilateral microtia-atresia. Address correspondence and reprint requests to Xiaowei Chen, M.D., Department of Otolaryngology, Peking Union Medical College Hospital, #1 shuaifuyuan, Beijing 100730, China; E-mail: chenxw_pumch@163.com This work was supported by grant to X.C. from the General Programs of National Natural Science Foundation of China (81271053) and The National Key Research and Development Program of China (2016YFC0901501). The authors have received no payment in the preparation of this manuscript. The authors alone are responsible for the content and writing of this article. X.C. acted as head surgeon for the BCHIs performed in this study and collected data. X.F. analyzed data, composed the manuscript, and participated in some of the operations as an assistant. T.Y., X.N., Y.W., and Y.F. participated in some of the operations as assistants, and collected and analyzed data. All authors read and approved the final manuscript. The authors disclose no conflicts of interest. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal's Website (http://journals.lww.com/otology-neurotology). This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0 Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Anatomical Correlates and Surgical Considerations for Localized Therapeutic Hypothermia Application in Cochlear Implantation Surgery Hypothesis: Application of localized, mild therapeutic hypothermia during cochlear implantation (CI) surgery is feasible for residual hearing preservation. Background: CI surgery often results in a loss of residual hearing. In preclinical studies, local application of controlled, mild therapeutic hypothermia has shown promising results as a hearing preservation strategy. This study investigated a suitable surgical approach to deliver local hypothermia in patients utilizing anatomical and radiologic measurements and experimental measurements from cadaveric human temporal bones. Methods: Ten human cadaveric temporal bones were scanned with micro-computed tomography and anatomical features and measurements predicting round window (RW) visibility were characterized. For each bone, the standard facial recess and myringotomy approaches for delivery of hypothermia were developed. The St. Thomas Hospital (STH) classification was used to record degree of RW visibility with and without placement of custom hypothermia probe. Therapeutic hypothermia was delivered through both approaches and temperatures recorded at the RW, RW niche, over the lateral semicircular canal and the supero-lateral mastoid edge. Results: The average facial recess area was 13.87 ± 5.52 mm2. The introduction of the cooling probe through either approach did not impede visualization of the RW or cochleostomy as determined by STH grading. The average temperatures at RW using the FR approach reduced by 4.57 ± 1.68 °C for RW, while using the myringotomy approach reduced by 4.11 ± 0.98 °C for RW. Conclusion: Local application of therapeutic hypothermia is clinically feasible both through the facial recess and myringotomy approaches without limiting optimal surgical visualization. Address correspondence and reprint requests to Suhrud M. Rajguru, Ph.D., University of Miami Ear Institute, 1095 NW 14 Terrance, Lois Pope Life Center, Room 4-25, Miami, Florida 33134; E-mail: s.rajguru@miami.edu E.P. and A.V.E. have equal contribution. This work was supported by a Research Grant from Cochlear, R01 DC01379801A1 and a pilot award from National Center For Advancing Translational Sciences of the National Institutes of Health under Award Number UL1TR002736, Miami Clinical and Translational Science Institute. S.M.R. and C.K. are named inventors on intellectual property related to the design of hypothermia system and probe discussed here. The authors declare no competing financial interests related to the findings presented. This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0 Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Evaluation of the Effect of Diclofenac Sodium and 5-Fluourasil in a 3D Cholesteatoma Cell Culture Model Introduction: Middle ear cholesteatoma is a benign disease with invasive and destructive clinical behaviors. It increases the rate of both chronic otitis media complications and revision surgeries. The most effective treatment of middle ear cholesteatoma is surgical excision, and there is no medical treatment for this disease. Exploring new medical treatment options may help to create treatment alternatives instead of surgery. Materials and Methods: Required cholesteatoma tissues for cell culture were excised from 4 different participants who underwent surgery in our clinic and agreed to give tissue for the study. Cholesteatoma-derived keratinocytes and fibroblasts were cocultured in temperature-sensitive culture dishes to make a three-dimensional (3D) cholesteatoma model. Then, the effects of 1% and 2% diclofenac sodium on viability and cell proliferation rates were examined using WST-1 and annexin-V tests. Results: Cell viability and proliferation rates were found to be lower and apoptosis rates were higher in the diclofenac sodium group versus the negative and positive control groups. Conclusion: In this present study, we described a new 3D cholesteatoma cell culture model developed using cell sheet technology and demonstrated the efficacy of diclofenac sodium on cholesteatoma for the first time in the literature. It may be used in patients with chronic otitis media with cholesteatoma, but further studies investigating ototoxic and neurotoxic effects of this molecule are needed. Address correspondence and reprint requests to Ahmet Kara, Sakarya University Training and Research Hospital, 54000 Korucuk, Sakarya, Turkey. E-mail: doktor.kbb@hotmail.com Funding: This study has been funded by Turkey Scientific and Technological Research Center (project number: 216S961) (TUBITAK). Date of presentation at scientific meeting: AAO-HNSF Annual Meeting & OTO Experience, 7–10.10.2018. The research protocol was approved by the Kocaeli University Animal Ethics Committee (KU GOKAEK 2013). The authors report no conflicts of interest Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Hearing Preservation With the Use of Flex20 and Flex24 Electrodes in Patients With Partial Deafness Objective: To evaluate the impact of electrode length on hearing preservation (HP) in Partial Deafness Treatment–Electrical Complement (PDT-EC) subjects. Study Design: Retrospective case review. Setting: Tertiary referral center. Patients: Twenty-three PDT-EC patients (with preoperative air-conduction thresholds ≤30 dB up to 500 Hz) were divided into two groups: Flex20 electrode (Med-EL GmbH, Innsbruck, Austria) (12 patients) and Flex24 electrode (Med-EL GmbH, Innsbruck, Austria) (11 patients). Interventions: All participants were subjected to minimally invasive cochlear implantation using the round window approach. Main Outcome Measure(s): Pure tone audiometry (125–8000 Hz) was performed preoperatively and at 1, 6, 12, and 24 months postoperatively. HP was established using the HEARRING group formula. Speech understanding was assessed preoperatively and at 12 and 24 months postoperatively. Results: Analysis of HP for every individual indicates that more than half the patients with Flex20 and Flex24 had complete HP at 6 months follow-up. None of the patients from either group had complete loss of hearing. At activation, average air-conduction thresholds for low frequencies (125–500 Hz) were slightly better for the short electrode (M = 29.03) than for the long (M = 39.10) but the difference was not statistically significant (p = 0.067). The effect of electrode (Flex20 versus Flex24) was not significant in terms of pure tone audiometry and speech recognition at long-term follow-up. Conclusions: In the early postoperative period, complete HP was possible in a majority of patients from both groups, but slightly better HP outcomes were achieved by Flex20. In the long term, the length of the electrodes does not affect the degree of HP or speech understanding. Address correspondence and reprint requests to Piotr H. Skarzynski, Ph.D., M.D., M.S., Mokra 17 Street, 05-830 Kajetany, Poland; E-mail: p.skarzynski@ifps.org.pl No other benefits were received. The authors disclose no conflicts of interest. Supplemental digital content is available in the text. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal's Website (http://journals.lww.com/otology-neurotology). This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND), where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal. http://creativecommons.org/licenses/by-nc-nd/4.0 Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Validating a New Tablet-Based Tool in the Determination of Cochlear Implant Angular Insertion Depth Objective: The objective of this study is to determine the reliability of a new tablet-based software that utilizes postoperative computed tomography to determine angular insertion depth (AID), cochlear duct length (CDL), and the cochlear place frequency of individual electrodes in cochlear implant recipients. Patients: Twenty adult cochlear implant recipients with lateral-wall electrode arrays of varying lengths were included in the study. Intervention: Cochlear and electrode array measurements were made by 2 otolaryngologists using a tablet-based software. The user manually identifies the modiolus, round window, and each electrode contact to calculate AID. The user also manually identifies cochlear landmarks to calculate the CDL. The AID and CDL are applied to the Greenwood function to obtain an estimate of the cochlear place frequency for each electrode. Main Outcome Measure(s): The primary outcome measure was the reliability of the instrument, as assessed with intra and interrater reliability of measured AID and CDL. The resultant differences in the estimated cochlear place frequency of the most apical electrode were also evaluated. Results: A broad range of AIDs were observed (390°–659°). Intraclass correlation coefficients for intra (0.991) and interrater reliability (0.980) of AID of the most apical electrode contact were excellent. Intra (0.820) and interrater reliability (0.784) of CDL were also excellent. The estimated cochlear place frequency for the most apical electrode differed by an average of 6.7% (0–18.7%) across the 2 raters. Conclusion: There is excellent agreement amongst clinicians in the determination of AID and CDL, resulting in small changes in estimated cochlear place frequency of the most apical electrode using this new software. Address correspondence and reprint requests to Michael W. Canfarotta, M.D., Department of Otolaryngology/Head and Neck Surgery, 170 Manning Drive, CB 7070, Physicians Office Building, Room G-190, Chapel Hill, NC 27599-7070, USA. E-mail: michael.canfarotta@unchealth.unc.edu Financial Disclosures/Conflicts of Interest: This project was funded in part by the NIH through NIDCD (T32 DC005360). The senior authors, H.C.P., K.D.B., and B.P.O., serve on the surgical advisory board for MED-EL Corporation. M.T.D. is supported by a research grant from MED-EL Corporation. M.W.C. and E.B. declare that involvement in research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. Author Contributions: M.W.C., M.T.D., and B.P.O. designed experiments. Implanting surgeons were H.C.P., K.D.B., and B.P.O. M.W.C., M.T.D., E.B., and B.P.O. wrote the article, and all authors contributed significantly to analysis and revisions leading to its final form. Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Differentiation Between Eosinophilic Otitis Media and Otitis Media Associated With Eosinophilic Granulomatosis With Polyangiitis Objective: To perform comparisons and clarify differences in clinical manifestations between eosinophilic otitis media (EOM) and otitis media associated with eosinophilic granulomatosis with polyangiitis (EGPA). Study Design: Retrospective case review. Setting: Tertiary referral center. Patients: Twenty-two ears of 11 patients exhibiting EOM (EOM group) and 20 ears of 12 patients exhibiting otitis media associated with EGPA (EGPA group). Main Outcome Measures: Otological manifestations, nasal and paranasal manifestations, incidence of asthma, positivity for serum antineutrophil cytoplasmic antibodies (ANCA), total serum immunoglobulin (Ig) E level, peripheral blood eosinophil fraction, and hearing outcomes. Results: The incidence and age of onset of asthma and chronic rhinosinusitis were comparable between the EOM and EGPA groups. Moreover, otological findings and hearing outcomes at the initial visit were similar in both groups. Computed tomography images of the paranasal sinus showed predominant opacification of the ethmoid sinus in both groups. Although the total serum IgE level was not significantly different, the peripheral blood eosinophil fraction was significantly larger in the EGPA group than in the EOM group (p = 0.0035). Furthermore, the rate of myeloperoxidase–antineutrophil cytoplasmic antibodies (ANCA) positivity was significantly higher in the EGPA group than in the EOM group (p = 0.019). Conclusions: The findings of the present study suggest that the phenotypic characteristics of EOM closely resemble those of otitis media associated with EGPA in early stages before the appearance of vasculitis. Therefore, it is challenging to differentiate the two conditions purely on the basis of otorhinological examinations. Address correspondence and reprint requests to Atsushi Fukuda, M.D., Department of Otolaryngology–Head & Neck Surgery, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, North 15, West 7, Kita-ku, Sapporo, Hokkaido 060-8638, Japan; E-mail: atsushi.fukuda@huhp.hokudai.ac.jp The authors do not have any conflicts of interest to declare. Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Characteristics and Outcomes of Pediatric Vestibular Schwannomas Objective: To review the demographics, treatment modalities, and survival of children with vestibular schwannomas. Study Design: Analysis using the Surveillance, Epidemiology, and End Results (SEER) database. Subjects and Methods: Pediatric patients from birth to 18 years in the SEER database were included from 2004 to 2014 based on a diagnosis of vestibular schwannoma using the primary site International Classification of Diseases (ICD) O-3 code of C72.4: acoustic nerve and the ICD O-3 histology codes of 9540/1: neurofibromatosis, Not Otherwise Specified (NOS); 9560/0: neurilemoma, NOS; or 9570/0: neuroma, NOS. Results: One hundred forty-eight pediatric vestibular schwannomas (VSs) cases were identified. The mean age at diagnosis was 13.9 years (range, 4.0–18.0). Eighty-five (57.4%) patients were women. Seventy-seven (52.0%) patients had isolated unilateral VSs while 71 (48.0%) patients had either bilateral VSs or unilateral VSs with other brain, spinal cord, or cranial nerve tumors. Eighty two (55.4%) patients received surgical resection only, 45 (30.4%) received no treatment, 6 (4.1%) received radiation only, and 12 (8.1%) received surgery and radiation. The median tumor size for patients who received no treatment was 9.5 mm (interquartile range [IQR]: 8.0) compared with 33.5 mm (IQR: 23.0) for patients who received surgical care and 41.0 mm (IQR: 1.5) for patients who received both surgery and radiation (p < 0.001). The 5-year overall survival rate was 97%. Conclusion: Pediatric VSs tend to be diagnosed in adolescence. No men or women predominance was appreciated. Treatment varied according to tumor size. Survival rates for children with vestibular schwannomas are excellent. These data may assist healthcare providers when counseling children with vestibular schwannomas and their families. Address correspondence and reprint requests to Tyler A. Janz, M.D., University of Central Florida College of Medicine, Orlando, FL; E-mail: tjanz@knights.ucf.edu This is the first submission on this topic. The authors have no funding, financial relationships, or conflicts of interest to disclose. Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

High-dose Corticosteroids for Adult Bell's Palsy: Systematic Review and Meta-analysis Objectives: To compare the efficacy and safety of high-dose corticosteroids (initial prednisolone [PSL] of 100 mg or more daily) and standard-dose corticosteroids (initial PSL of 50–60 mg) in patients with Bell's palsy. Study Design: A systematic review and meta-analysis. Data Sources: Medline, Embase, Cochrane Central Register of Controlled Trials, Ichushi-Web, Web of Science, and CINAHL, combined with data from ClinicalTrials.gov. Study Selection: Published and unpublished cohort studies comparing high- and standard-dose corticosteroids in adult patients with Bell's palsy were included. Data Extraction: Study characteristics (study design, patient's number), patient characteristics (sex, age, disease severity, prescription of antivirals), and outcomes (nonrecovery, any adverse effects). Data Synthesis: From the 1,974 identified articles, 8 studies were met eligible criteria. Of the included studies, the initial dose in high-dose corticosteroids regimens varied from 120 mg to 200 mg PSL daily. Compared with standard-dose corticosteroids, high-dose corticosteroids were associated with a significantly decreased nonrecovery at 6 months after disease onset (odds ratio 0.42, 95% confidence interval 0.22–0.80; very low quality) in patients with Bell's palsy. No severe adverse effects were observed in patients receiving high- or standard-dose corticosteroids. Conclusion: High-dose corticosteroids reduce nonrecovery in patients with Bell's palsy. The dose of high-dose corticosteroids was varied and further prospective study is needed to identify an adequate dose of corticosteroids in these patients. Address correspondence and reprint requests to Takashi Fujiwara, M.D., Ph.D., Department of Otolaryngology Head and Neck Surgery, Kurashiki Central Hospital, 1-1-1 Miwa, Kurahiki, Okayama 710-8602, Japan; E-mail: t.fujiwarabi@gmail.com All authors declare: no support from any organization for the submitted work; no financial relationships with any organizations that might have an interest in the submitted work in the previous 3 years; no other relationships or activities that could appear to have influenced the submitted work. The authors disclose no conflicts of interest. Supplemental digital content is available in the text. Supplemental digital content is available for this article. Direct URL citations appear in the printed text and are provided in the HTML and PDF versions of this article on the journal's Website (www.jcraniofacialsurgery.com). Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Endre Hőgyes (1847–1906), Forgotten Father of the Vestibulo-Ocular Reflex Objective: Throughout the history of vestibular research, the discovery of the vestibulo-ocular reflex in 1881 by Endre Hőgyes (1847–1906) is rarely mentioned. The aim of this study is to review Hőgyes' vestibular research articles, all originally written in Hungarian and emphasize their epoch-making content. Main Data Sources: Hőgyes' vestibular publications, originally written in Hungarian, which describe various eye movements of the rabbit in response to vestibular stimulation by rotation about three axes. Results: Hőgyes was the first to use a three-axis turntable on an experimental animal, in this case a rabbit. He found that depending on the plane of rotation, different types of binocular eye movements were produced. He then demonstrated by destructive and excitatory experiments, the anatomical pathways and the physiological function producing this phenomenon. Ultimately, he explained the exact connections between the inner ear labyrinth and certain muscle contractions during eye movements. He identified this pathway as the "associating center of the ocular movements," later defined as the vestibulo-ocular reflex. Hőgyes' discovery was only superficially noted during his lifetime and ignored after his death. Conclusion: Hőgyes was the first to demonstrate the vestibulo-ocular reflex. He was forgotten during the ensuing 140 years probably because his articles were appeared only in Hungarian and because a short time later, Róbert Bárány's award of the Nobel Prize overshadowed many of Bárány's predecessors and contemporaries, including Hőgyes and relegated them to the background. Address correspondence and reprint requests to László T. Tamás, M.D., 9028, Győr, Vak Bottyán u.14/A, Hungary; E-mail: ltamas@gyor.net Dr. L.T.T. and Dr. A.M. helped in data collection, analysis, and interpretation, critically reviewing and editing the manuscript. The authors disclose no conflicts of interest. Copyright © 2019 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Alexandros Sfakianakis
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