Otology & Neurotology

Predictive Ability of Direct Electrical Stimulation on Facial Nerve Function Following Vestibular Schwannoma Surgery: A Systematic Review and Meta-analysis Objective: Facial nerve preservation is critical in vestibular schwannoma (VS) surgery. Direct electrical stimulation (DES) is a widely used method for intraoperative localization of the facial nerve and assessment of nerve integrity. We sought to assess the predictive ability of DES parameters on facial nerve function post-VS surgery. Databases reviewed: A systematic literature search of English-language studies published from 1946 to 2019 was undertaken using EMBASE, MedLine/PubMed, and the Cochrane Central Register of Controlled Trials. Methods: Included studies involved patients undergoing VS surgery and assessed predictive ability of any DES parameter on postoperative facial function. Two authors independently reviewed studies and extracted data. Meta-analysis of diagnostic accuracy of DES parameters was used to calculate pooled sensitivities and specificities of common cutoffs. For DES parameters reported by less than four studies, outcomes were reported descriptively. Results: A MST cutoff of 0.10 mA had sensitivity of 0.76 (95% CI 0.53–0.90) and specificity 0.68 (95% CI 0.42–0.87) for facial function in the long term, and MST 0.05 mA had sensitivity 0.73 (95% CI 0.58–0.84) and specificity 0.74 (95% CI 0.59–0.85). CMAP amplitude < 500 μV had sensitivity of 0.87 (95% CI 0.78–0.93) for poor short-term facial function. Conclusions: A CMAP amplitude response < 500 μV is sensitive for poor short-term facial function, whereas MST values of 0.05 mA and 0.10 mA are both sensitive and specific in the long term. Other DES parameters may be able to accurately predict both short- and long-term postoperative facial function, but have been less studied. Address correspondence and reprint requests to Dr. Joseph Chen, M.D., F.R.C.S.C., Department of Otolaryngology–Head and Neck Surgery, Sunnybrook Health Sciences Centre, University of Toronto, 2075 Bayview Ave., Room M1 102, Toronto, ON M4S 3M5, Canada. E-mail: aquim047@uottawa.ca 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). Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Pulsatile Tinnitus in Superior Semicircular Canal Dehiscence Cured by Endovascular Coil Occlusion of the Superior Petrosal Sinus No abstract available

When to Refer a Hearing-impaired Patient for a Cochlear Implant Evaluation Objectives: To explore the predictive value of utilizing routine audiometry to best determine cochlear implant (CI) candidacy using AzBio sentences. Methods: A retrospective chart review was performed between 2011 and 2018 for 206 adult patients who underwent CI evaluation assessed with AzBio sentences. Better hearing ear word recognition score (WRS) using Northwestern University-6 word lists presented at decibel hearing level from a standard audiogram was used to determine when best to refer a patient for CI evaluation. Predicted AzBio scores from multivariate regression models were calculated and compared with the actual CI candidacy to assess accuracy of the regression models. Results: Race, marital status, hearing aid type, better hearing ear WRS, and HL were all independently and significantly associated with AzBio testing in quiet on univariate analyses. Better hearing ear WRS and better hearing ear decibel hearing level predicted AzBio Quiet on multivariate regression analysis. For AzBio +10 dB signal-to-noise ratio (SNR), sex, and better hearing ear WRS each significantly predicted speech perception testing. Predicted CI candidacy was based on AzBio sentence testing of ≤60% for the ease of statistical analysis. Regression models for AzBio sentence testing in quiet and +10 dB SNR agreed with the actual testing most of the time (85.0 and 87.9%, respectively). A generalized linear model was built for both AzBio testing in quiet and +10 dB SNR. Conclusion: A WRS of <60% in the better hearing ear derived from a routine audiogram will identify 83.1% of CI candidates while appropriately excluding 63.8% of patients. Address correspondence and reprint requests to Jacob B. Hunter, M.D., Department of Otolaryngology—Head and Neck Surgery, University of Texas Southwestern Medical Center, 2001 Inwood Road, Dallas, TX 75235; E-mail: jacob.hunter@utsouthwestern.edu Institutional Review Board Approval(s): STU 032018-085. Internal departmental funding was utilized without commercial sponsorship or support. The views expressed in this manuscript are those of the author(s) and do not reflect the official policy or position of the Department of the Army, Department of Defense, or the US Government. The authors disclose no conflicts of interest. Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

The Relationship Between Otitis Media With Effusion and Gastroesophageal Reflux Disease: A Meta-analysis Objective: Recent studies have investigated the mechanism by which refluxed gastric materials reach the middle ear, to establish otitis media with effusion (OME) causal relation between them in both children and adults. Therefore, the relationship between OME and gastro-esophageal reflux disease (GERD) should be further studied extensively. Methods: To identify eligible original articles, we searched a range of computerized databases, including Medline via PubMed, EMBASE, CNKI, and Web of Science with a systematic searching strategy. Subgroup analysis was performed to analyze heterogeneity and Egger and Begg funnel plot to assess the publication bias of the included articles. Results: The meta-analysis had an overall sample size of 1961. We identified a significant relationship between OME and GERD, with a pooled odds ratio (OR) of 4.52 (95% confidence interval [CI]: 2.42–8.44; p < 0.001). The pooled data were calculated with the random-effects model as a high significant heterogeneity was found among the studies and there was no significant publication bias observed. Conclusions: The meta-analysis suggested that there was a significant association between otitis media with effusion and gastroesophageal reflux disease. Address correspondence and reprint requests to Hai-Ying Sun, M.D., Department of Otorhinolaryngology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei, China; E-mail: sunhaiying120@163.com Z.-H.W., Y.T., and X.N. are joint first author. These authors contributed equally to this study. H-Y.S. and X.C. are joint corresponding authors. Author Contributions: All authors contributed significantly to this work. W.Z.H. and T.Y. designed the research study; W.Z.H. and N.X performed the research study and extracted the data and analyzed the data; W.Z.H., N.X., S.H.Y. and C.X. wrote and revised the manuscript. In addition, all authors approved the final draft. Competing financial interests: No financial (no Funding, Employment and Personal financial interests) and Non-financial competing interests. This work is supported by grants from The National Natural Science Foundation of China (81600801). The authors disclose no conflicts of interest. Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Intracanalicular Vestibular Schwannoma: A Systematic Review and Meta-analysis of Therapeutics Outcomes Objective: To perform a systematic review and meta-analysis summarizing the current evidence on the management of intracanalicular vestibular schwannoma. Data Sources: Embase (1947–), Medline (1946–), Cochrane library (1947–), Scopus (2010–), and CINAHL (1961–) were searched from 1969 to October 5, 2019 (50 years). Study Selection: A search strategy was performed to identify patients with vestibular schwannoma confined to the internal auditory canal without extension to the cerebellopontine angle. Studies with patients aged less than 18, Neurofibromatosis type 2, revision cases, and non-English language were excluded. Data Extraction: A standardized collection sheet was used for the extracted data and a quality assessment was performed using the Newcastle-Ottawa Scale with the comparability criterion omitted. Data Synthesis: Seventy-one studies were included with 24 on observation, 14 on radiotherapy, and 34 on surgery. The primary outcome was serviceable hearing preservation. Secondary outcomes were preservation of facial nerve function, growth, involution, and dizziness. Sub-analysis on the type of surgery and type of radiotherapy were performed. Excel 2016 with MIX 2.0 Pro add-on package was used to analyze the data and create forest plots. Data were presented in proportion with a 95% confidence interval. Conclusions: Serviceable hearing was observed in 31% of patients after observation, 56% after radiotherapy, and 51% after surgical treatment with mean follow-up time of 4.04 years, 4.92 years, and 2.23 years, respectively. Facial nerve function was found to be best preserved in both observation and radiotherapy groups. Vestibular schwannoma growth occurred in 33% of patients under observation. Involution occurred in 2% of patients under observation and in 38% after radiotherapy. Address correspondence and reprint requests to Marina Neves Cavada, M.D., Sydney Adventist Hospital, 185 Fox Valley Rd, Wahroonga, Sydney, NSW 2076, Australia; E-mail: marinacavada@gmail.com Disclosure of funding: No funding or other supports were received. The author discloses 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). Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Hearing Outcomes With a Novel Total Ossicular Replacement Prosthesis Introduction: A total ossicular replacement prosthesis (TORP) is used to reconstruct the ossicular chain in the absence of the stapes suprastructure. The Wildcat prosthesis is a novel TORP that eliminates the need for a separate footplate shoe prosthesis and aims to improve ease-of-use and stability. This study evaluates hearing outcomes using the Wildcat prosthesis. Study Design: Case series with chart review. Setting: Tertiary neurotology referral center. Methods: Retrospective chart review of 64 patients undergoing ossicular chain reconstruction using the Wildcat TORP. Hearing outcomes after surgery were assessed with air conduction pure-tone average, bone conduction pure-tone average, air-bone gap (ABG), speech recognition threshold , and word recognition score as primary outcome measures. The stability of hearing outcomes was evaluated on subsequent long-term follow-up. Results: At mean short-term follow-up of 4.4 ± 2.7 months, ABG improved from 31.0 ± 13.0 dB preoperatively to 22.5 ± 10.0 dB (p < 0.001) with 51.6% achieving ABG less than 20 dB. No significant difference in any primary outcome measures was found when analyzing outcomes by initial versus revision surgery, use of cartilage graft, or type of mastoidectomy. The only exception was a smaller reduction in ABG of 4.2 dB for patients with canal wall down mastoidectomy compared with a 13.7 dB ABG closure in patients with canal wall up mastoidectomy (p = 0.039). Conclusion: Total ossicular chain reconstruction using the Wildcat demonstrates versatility in challenging cases to provide hearing outcomes that are comparable to published data using TORPs. Address correspondence and reprint requests to Kenny F. Lin, M.D., Michigan Ear Institute, 30055 Northwestern Highway, #101, Farmington Hills, MI 48334; E-mail: kenny.f.lin@gmail.com All authors have approved the final manuscript and attest to the integrity of the original data and the analysis reported in the manuscript. The authors disclose no conflicts of interest. Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Which Inner Ear Disorders Lie Behind a Selective Posterior Semicircular Canal Hypofunction on Video Head Impulse Test? Objective: To assess all different patterns of associated abnormalities on audiometry, bithermal caloric test (BCT) and cervical/ocular vestibular-evoked myogenic potentials (VEMPs) to air/bone-conduction in patients with selective posterior semicircular canal (PSC) hypofunction and to correlate them with underlying disorders. Study Design: Retrospective review. Setting: Tertiary referral center. Patients: 51 patients (23 men, 28 women, mean age 57.5 yr) with isolated PSC deficit (one bilateral). Interventions: Correlation with instrumental data and underlying diagnoses. Main Outcome Measures: Video-oculographic findings, objective measurements on audiometry, BCT, VEMPs and video-head impulse test (vHIT). Results: Ongoing or previous acute vestibular loss (AVL) was diagnosed in 13 patients (25.5%, 3 inferior vestibular neuritis, 10 AVL with sudden sensorineural hearing loss [SSNHL]), Meniere's disease (MD) in 12 (23.5%), cerebellopontine angle (CPA) lesion in 9 (17.6%), various causes in 7 (13.7%), benign paroxysmal positional vertigo (BPPV) involving the non-ampullary arm of PSC in 5 cases (9.8%) whereas unknown pathology in 5 (9.8%). Involvement of at least one additional receptor besides PSC was seen in 89.8% of cases. Cochlear involvement was diagnosed in 74.5% with pure-tone average significantly greater in patients with AVL+SSNHL (p < 0.05). Overall involvement of labyrinthine receptors or afferents was highest in patients with AVL+SSNHL (p < 0.01), MD and CPA lesions (p < 0.05). Conclusions: Isolated loss of PSC function on vHIT is mostly accompanied by additional labyrinthine deficits that could only be identified through an accurate instrumental evaluation. Assessment of all receptors and afferents should be always pursued to identify the lesion site and better understand the underlying pathophysiological mechanisms. Address correspondence and reprint requests to Andrea Castellucci, M.D., ENT Unit, Department of Surgery, Arcispedale Santa Maria Nuova, AUSL - IRCCS, Viale Risorgimento 80, 42123 Reggio Emilia, Italy; E-mail: andrea.castellucci@ausl.re.it Sources of support that require acknowledgment: none declared. Source of funding: none declared. Ethical statement: All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee (Ethics Committee of our Institution, approval number 273/2017/O/OssN) and with World Medical Association Declaration of Helsinki (2002). Informed consent was obtained from all individual participants included in the study. 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). Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Comparative Performance of Lateral Wall and Perimodiolar Cochlear Implant Arrays Objective: The physical shape of cochlear implant (CI) arrays may impact hearing outcomes. The goal of this study was to compare post-operative speech and melody perception between patients with lateral wall (LW) and perimodiolar (PM) electrode arrays across a range of lengths and manufacturers. Study Design: Retrospective chart review. Setting: Tertiary Care Hospital. Patients: 119 adult patients with post-lingual hearing loss who underwent cochlear implantation. Main Outcome Measures: A total of seven different electrodes were evaluated including 5 different LW electrodes (CI422 [Cochlear American], 1J [Advanced Bionics], Medium [Med El], Standard [Med El], Flex28 [Med El]) and 2 PM electrodes (Contour [Cochlear American], MidScala [Advanced Bionics]). Speech perception outcomes (n = 119 patients) were measured by Consonant-Nucleus-Consonant (CNC) scores collected 3, 6, 12 and 24 months after implantation. Melody perception outcomes (n = 35 CI patients and n = 6 normal hearing patients) were measured by Melodic Contour Identification (MCI). Results: CNC scores increased over time after implantation across all array designs. PM designs exhibited higher CNC scores compared to LW electrodes, particularly 6-months after implantation. Pre-operative pure tone averages did not correlate with post-operative CNC scores. PM arrays outperformed LW electrodes in terms of MCI scores. Conclusions: The physical shape of cochlear implant electrode arrays may impact hearing performance. Compared to LW designs, PM arrays appear to offer superior speech perception during the first 6 months after implantation, with performance equalizing between groups by 24 months. Compared to LW designs, PM arrays also appear to afford superior melody perception. Correspondence: Ana H. Kim, M.D., Division of Otology, Neurotology and Skull Base Surgery, Department of Otolaryngology–Head and Neck Surgery, Columbia University Irving Medical Center, New York, NY; E-mail: ahk2166@cumc.columbia.edu Funding and Conflict of Interest: M.K. served on the advisory board for Med El. A.H.K. receives research funding from Advanced Bionics. No funding was received from either company or any other entity to support this project. 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). Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

Is Menière's Disease a Contraindication to Stapedectomy? Objective: Review surgical outcomes of stapedectomy for otosclerosis in patients with Menierè's disease. Study Design: Retrospective case review. Setting: Tertiary referral center. Patients: Patients with otosclerosis and Menière's disease undergoing stapedectomy between 2010 and 2017. Intervention: Stapedectomy. Main Outcome Measures: Pre- and postoperative hearing and complications. Hearing was measured by air conduction (AC) and bone conduction (BC) pure-tone frequency, pure-tone averages (PTA), air-bone gap (ABG), and word recognition scores (WRS). Results: Among 1,499 patients with otosclerosis, the incidence of concomitant Menière's disease was 1.7%. Fifteen patients with otosclerosis and Menière's disease underwent stapedectomy, 12 primary and three revisions. Mean AC PTA was 43 dB preoperatively, and 25 dB postoperatively (p = 0.0007), while the ABG improved on average from 20 to 5 dB (p = 0.0001). There was no significant difference in BC PTA or WRS postoperatively. Two patients experienced fluctuation of hearing in the postoperative period, one of which resolved with a course of steroids. The mean follow-up time was 41 months. Conclusions: In patients with otosclerosis and Menière's disease, stapedectomy provides excellent hearing outcomes in a majority of patients. As is characteristic of Menière's disease, some patients will continue to experience fluctuating hearing postoperatively, which may progress to severe sensorineural hearing loss. Menière's disease may not be an absolute contraindication to stapes surgery. Address correspondence and reprint requests to Pedrom C. Sioshansi, M.D., Michigan Ear Institute, 30055 Northwestern Highway, Suite 101, Farmington Hills, MI 48334; E-mail: psioshansi@gmail.com The authors disclose no conflicts of interest. Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

A Novel COCH Mutation Affects the vWFA2 Domain and Leads to a Relatively Mild DFNA9 Phenotype Objective: To study the genotype and phenotype of a Dutch family with autosomal dominantly inherited hearing loss. Study Design: Genotype–phenotype correlation study. Genetic analysis consisted of linkage analysis, variable number of tandem repeats analysis, and Sanger sequencing. Audiovestibular function was examined. Regression analysis was performed on pure tone audiometry and speech recognition scores and correlated with the age and/or level of hearing loss. Setting: Tertiary referral center. Patients: A large Dutch family presenting with sensorineural hearing loss. Main Outcome Measures: Identification of the underlying genetic defect of the hearing loss in this family. Results of pure tone and speech audiometry, onset age, progression of hearing loss and vestibular (dys)function. Results: A novel mutation in COCH, c.1312C > T p.(Arg438Cys), cosegregates with hearing loss and a variable degree of vestibular (dys)function in this family. The reported mean age of onset of hearing loss is 33 years (range, 18–49 yr). Hearing loss primarily affects higher frequencies and its progression is relatively mild (0.8 dB/yr). Speech perception is remarkably well preserved in affected family members when compared with other DFNA9 families with different COCH mutations. Conclusion: These findings expand the genotypic and phenotypic spectrum of DFNA9. The c.1312C > T mutation, which affects the vWFA2 domain, causes a relatively mild audiovestibular phenotype when compared with other COCH mutations. Address correspondence and reprint requests to Ronald J. E. Pennings, M.D., Ph.D., Department of Otorhinolaryngology, Hearing & Genes, Radboud University Medical Center, Internal postal code 377, P.O. Box 9101, 6500 HB Nijmegen, the Netherlands; E-mail: Ronald.Pennings@radboudumc.nl H.K. and E.V. have contributed equally to this article. This work was financially supported by a grant from the Heinsius Houbolt foundation [to H.K. and R.J.E.P.]. The authors disclose no conflicts of interest. Web resources: Deafness Variation Database (http://deafnessvariationdatabase.org/) (Accessed: 13-08-2020). ClinVar (https://www.ncbi.nlm.nih.gov/clinvar/ (Accessed: 13-08-2020). 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). Copyright © 2020 by Otology & Neurotology, Inc. Image copyright © 2010 Wolters Kluwer Health/Anatomical Chart Company

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