Publications by authors named "Ryoukichi Ikeda"

58 Publications

Patulous Eustachian Tube Patients with Respiratory Fluctuation of Tympanic Membrane in Both Sitting and Supine Positions: A Sign of Severity of Disease?

Otol Neurotol 2021 May 12. Epub 2021 May 12.

Sen-En Rifu Otological Surgery Center, Miyagi Department of Otolaryngology-Head and Neck Surgery, Tohoku University School of Medicine, Sendai, Japan.

Objective: The aim is to report four cases of patulous Eustachian tube (PET) patients with respiratory fluctuation of the tympanic membrane (TM) even in the supine position, and to examine the frequency and characteristics of such patients.

Patients: There were 195 ears (99 right ears and 96 left ears) from 146 cases (56 male and 90 female subjects aged 8-88, average 48.0 ± 18.9 yrs) diagnosed with definite PET by diagnostic criteria proposed by Japan Otologic Society (JOS) between January 2017 and December 2019 at Sen-En Rifu Hospital. Patients who presented with respiratory fluctuation of the TM in both the sitting and supine positions were examined.

Main Outcome Measures: Clinical patient records, the severity of subjective symptoms (PET handicap inventory-10 [PHI-10]), objective ET function tests (tubo-tympano-aerodynamic graphy [TTAG] and sonotubometry), and sitting three-dimensional computed tomography (3-D CT) were analyzed.

Results: Six ears (3.1%) of four cases (2.7%) exhibited respiratory fluctuation of the TM, even in the supine position. In these six ears (four cases), the PHI-10 score ranged from 16 to 36 with three ears exhibiting PHI-10 score equal to or exceeding 26 (in the category of severe handicap). Sitting CT indicated the findings of completely open ET in only two ears. All ears but one were managed by conservative treatment.

Conclusion: Respiratory fluctuation of the TM in both the sitting and supine positions was observed in 2.7% of the definite PET patients. Surprisingly, such findings can be an indication of neither the subjective nor objective severity of the disease. Therefore, even for PET patients with such findings, surgery should not be immediately proposed, but rather, conservative management should be undertaken first.
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http://dx.doi.org/10.1097/MAO.0000000000003190DOI Listing
May 2021

A Novel Mutation in LMX1B (p.Pro219Ala) Causes Focal Segmental Glomerulosclerosis with Alport Syndrome-like Phenotype.

Intern Med 2021 Apr 5. Epub 2021 Apr 5.

Division of Nephrology, Endocrinology, and Vascular Medicine, Tohoku University Graduate School of Medicine, Japan.

A 69-year-old woman presented with mild renal dysfunction, proteinuria, and sensorineural hearing loss. A renal biopsy showed focal segmental glomerulosclerosis with thinning of the glomerular basement membrane. There was a positive family history of end-stage kidney disease and hearing loss. Although Alport syndrome was suspected from these features, a genetic test using next-generation sequencer identified a novel missense mutation in LMX1B, c.655C>G: p. (Pro219Ala). In silico analyses predicted the pathogenicity of the mutation. Thus, the present case was diagnosed as LMX1B-associated nephropathy presenting with Alport syndrome-like phenotype, expanding the disease spectrum of LMX1B nephropathy.
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http://dx.doi.org/10.2169/internalmedicine.6987-20DOI Listing
April 2021

Questionnaire survey on pharyngolaryngeal sensation evaluation regarding dysphagia in Japan.

Auris Nasus Larynx 2021 Aug 15;48(4):666-671. Epub 2021 Feb 15.

Department of Otolaryngology and Head and Neck Surgery, Tohoku University Graduate School of Medicine. Electronic address:

Objective: The objective is to conduct a questionnaire survey regarding pharyngolaryngeal sensation evaluation in dysphagia to understand the current situation in Japan.

Method: The questionnaire was sent to the councilor of the Society of Swallowing and Dysphagia of Japan and the Japanese Society of Dysphagia Rehabilitation-Certified Clinician. The prospective questionnaire survey included the questions listed below: Q1: What do you think of the importance of pharyngolaryngeal sensory evaluation? Q2: Select one of the essential swallowing sensations. Q3: Select one of the following regarding the frequency of sensory examination of the larynx. Q4: Select the proportion of cases the sensory test results affect. Q5: As a pharyngolaryngeal sensory evaluation method in swallowing function evaluation, please fill in the table below for the frequency, difficulty, and effectiveness of the following tests, such as gag reflex, touching the larynx by endoscopy, touching the larynx by the probe with endoscopy, cough reflex test, swallowing provocation test.

Results: The essential swallowing sensations of mechanical stimulation, chemical stimulation, thermal stimulation were 84.9%, 5.4%, and 9.7%, respectively. The frequency of touching the larynx by endoscopy in the otolaryngology group and cough reflex test in dentistry was significantly higher than the other groups (p < 0.05). The correlation between the frequency and difficulty or effectiveness of the sensory tests indicated that the frequency and difficulty are significantly correlated between each item.

Conclusion: Our results aid in increasing understanding and selection of pharyngolaryngeal sensation evaluation for dysphagia patients.
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http://dx.doi.org/10.1016/j.anl.2021.01.022DOI Listing
August 2021

Loudness functions for patients with functional hearing loss.

Int J Audiol 2021 Feb 15:1-7. Epub 2021 Feb 15.

Department of Audiology, Tohoku University Graduate School of Medicine, Sendai, Japan.

Objectives: To compare the loudness functions (loudness ratings as a function of sound level) obtained from patients diagnosed as having functional hearing loss (FHL) with those for patients with sensorineural hearing loss (SNHL) and healthy volunteers.

Design: Loudness functions for a 1000 Hz tone for patients with FHL and SNHL were assessed based on the categorical loudness scaling method. The data were compared with control data obtained in our facilities.

Study Sample: 18 patients (33 ears) with FHL and 10 patients (19 ears) with SNHL.

Results: For patients with SNHL and healthy volunteers, loudness increased progressively with increasing sound level above the audiometric threshold, with no exceptions. However, for about 70% of the patients with FHL, a different type of loudness function was obtained; the thresholds determined from the loudness function, which were defined as the minimum sound levels at which loudness could be judged, were 10 dB or more lower than the audiometric threshold (>10 dB), and/or the loudness ratings were elevated for a sound at the audiometric threshold.

Conclusions: The results support the hypothesis that patients with FHL often make threshold judgments based on a certain loudness.
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http://dx.doi.org/10.1080/14992027.2021.1881627DOI Listing
February 2021

Objective assessment of autophony during phonation in the diagnosis of patulous Eustachian tube patients.

Auris Nasus Larynx 2021 Aug 29;48(4):738-744. Epub 2020 Dec 29.

Sen-En Rifu Otological Surgery Center, Miyagi, Japan.

Objective: A system enabling the objective assessment of the transmission of voice sounds to the external auditory canal (EAC) during phonation has recently been revised. Our aim was to evaluate the effectiveness of this new system in the diagnosis of patulous Eustachian tube (PET) patients by comparing the results obtained using this method with those obtained from conventional objective tests to diagnose PET.

Methods: A prospective survey of medical records was included with definite PET, possible PET, and sensorineural hearing loss as control. The measurement system consists of a personal computer, an AD/DA converter (NI 6361, National Instruments), a probe microphone system for recording voice sound (ER-10C, Etymotic Research) and two microphones for measuring noise sound in the EAC (ER-10B+, Etymotic Research). Pronouncing the "Ni" sound for 5 s were recorded with these three microphones. The ratio of the maximum sound pressure of voice sound and noise sound in EAC (EAC/Voice) was simultaneously calculated, and results were displayed on a personal computer for diagnosing.

Results: Thirty-one patients of 42 ears with definite PET, 26 patients of 38 ears with possible PET, and 12 patients of 24 ears with sensorineural hearing loss as control were included. The EAC/Voice were 8.63 ± 5.43, 25.41 ± 32.63, and 25.87 ± 24.93 in the control, definite PET, and possible PET group respectively. The control group was significantly different from the definite PET (p < 0.05) and possible PET group (p < 0.05). ROC curve analysis confirmed 14.7 as the best diagnostic cut-off value of EAC/Voice (area under the curve=0.782, 95% CI 0.671-0.894). By adopting this cut-off point, 25 (56.8%) and 22 (61.1%) ears were determined as positive findings in the definite PET and possible PET group, respectively. There was no significant correlation between the positive findings judged by the current method and that of sonotubometry in the control (r = -0.63, p = 0.769), definite PET (r = 0.12, p = 0.451), and possible PET group (r = 0.12, p = 0.451).

Conclusion: The current system is more useful in the objective assessment of autophony during phonation by calculating the ratio of voice sound and elicited noise sound transmitted in the EAC (EAC/Voice). This method seems promising because it is able to detect cases eluding conventionally used test methods such as sonotubometry performed without phonation, thereby increasing the accuracy of PET diagnoses.
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http://dx.doi.org/10.1016/j.anl.2020.12.001DOI Listing
August 2021

Middle ear adenoma with facial palsy: A case report and a review of the literature: Middle ear adenoma with facial palsy.

Auris Nasus Larynx 2020 Nov 24. Epub 2020 Nov 24.

Department of Otolaryngology and Head and Neck Surgery, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai 980-8574, Japan. Electronic address:

A 52-year-old man presented to our emergency department with an acute onset of right-sided facial nerve (FN) palsy of House-Brackmann grade V. Electroneurography (ENoG) was conducted with no response at the right FN, as compared with the left FN (0%). We performed a biopsy of the right middle ear mass and histological studies showed the tumor to be neuroendocrine tumors (NET) of the middle ear. We resected the tumor with canal wall down mastoidectomy and reconstructed the posterior meatal wall with soft tissue. Three months after surgery, the FN paralysis had improved with House-Brackmann grade II. We reviewed cases of NET with FN palsy, and nine patients, including our case, have been reported. Our case is the first report of ENoG for the description of FN palsy due to NET. Although the ENoG value was 0%, it was remarkably improved by surgery. The other cases of NET patients with FN palsy also recovered FN function after surgery. These results suggest that it is recommended to perform the total resection of the tumor to improve the FN function.
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http://dx.doi.org/10.1016/j.anl.2020.11.003DOI Listing
November 2020

Systematic Review of Surgical Outcomes Following Repair of Patulous Eustachian Tube.

Otol Neurotol 2020 09;41(8):1012-1020

Sen-En Rifu Otologic Surgery Center.

Objective: To perform a systematic review and meta-analysis of surgical outcomes following repair of patulous Eustachian tube (PET).

Data Sources: Analysis of cases collected from studies published between January 1990 and December 2018 and identified using PubMed, Google Scholar, and Cochrane databases.

Study Selection: Articles addressing interventions to treat PET were selected.

Data Extraction: The database was searched using the keywords "patulous Eustachian tube treatment" and yielded 1,370 studies. Twenty studies were eligible for inclusion.

Data Synthesis: A total of 914 cases were evaluated for surgical approach, patient outcomes, and complications. The reported techniques were categorized by the procedure type, including ventilation tube insertion, mass loading of the tympanic membrane, ET injection, plug surgery, shim surgery, tuboplasty, and ET closure. Overall PET symptom improvement were reported for ventilation tube insertion (79 cases, mean: 58%, 95% CI: 47-69%), mass loading of the tympanic membrane (43 cases, mean: 50%, 95% CI: 32-69%), ET injection (139 cases, mean: 47%, 95% CI: 38-56%), plug surgery (386 cases, mean: 81%, 95% CI: 77-85%), shim surgery (122 cases, mean: 62%, 95% CI: 53-70%), tuboplasty (105 cases, mean: 41%, 95% CI: 31-51%), and ET closure (40 cases, mean: 66%, 95% CI: 49-80%). A low incidence of minor complications was reported.

Conclusions: This systematic review describes clinical outcome data following surgical management of PET. Plug surgery and shim surgery show relatively high effectiveness and safety. Further prospective studies that compare surgical approaches for PET are needed.
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http://dx.doi.org/10.1097/MAO.0000000000002753DOI Listing
September 2020

Management of Patulous Eustachian Tube.

JMA J 2020 Apr 6;3(2):101-108. Epub 2020 Apr 6.

Sen-En Rifu Otological Surgery Center, Miyagi, Japan.

Patients with patulous Eustachian tubes (PET) suffer from annoying aural symptoms, such as voice or breath autophony, and aural fullness due to the ET's abnormal patency. It may lead to an enormous reduction in quality of life. Various treatment methods, including conservative and surgical therapy, have been reported. In most cases, conservative treatment is sufficient to relieve patients of aural symptoms. However, some chronic and severe cases are resistant to traditional conservative therapy. Recently performed prospective and multicenter trials revealed the efficacy and safety of a silicone plug (Kobayashi plug) insertion for patients with severe PET. Patulous Eustachian tube handicap inventory-10 (PHI-10), tubal obstruction procedures, sitting computed tomography (CT), and ET function tests (tubo-tympano-aerodynamic graphy (TTAG) and sonotubometry) are useful for diagnosis as well as selecting candidates for surgery in severe cases.
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http://dx.doi.org/10.31662/jmaj.2020-0007DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7590401PMC
April 2020

A Facelift Procedure for Resection of Benign Parapharyngeal Tumors.

Otolaryngol Pol 2020 Jun;74(5):1-5

Department of Otolaryngology, Head and Neck Surgery, Toho University Faculty of Medicine, Tokyo, Japan.

<b>Objective:</b> The feasibility, surgical outcomes and possible risks and complications encountered during a facelift procedure for patients with parapharyngeal space (PPS) tumor were analyzed. <br><b>Method:</b> This retrospective analysis examined 10 patients who underwent surgery for PPS tumor using a facelift incision at our institutes between April 2015 and August 2019. <br><b>Results:</b> This study included four retro-styloid (benign nerve sheath tumor) and six pre-styloid tumors (pleomorphic adenoma). Mean tumor dimensions were 4.1 x 4.2 x 3.8 cm respectively. None of the patients needed conversion to conventional open resection. Transient sensory changes in the auricle occurred in 30% of the patients; however, all recovered within four months. In all the patients, postoperative scars were fully concealed by the auricle and hair. No recurrences were detected during a mean follow-up period of 16.6 months. <br><b>Conclusion: </b>The facelift procedure provides adequate visualization, workspace and excellent cosmetic results in properly selected cases.
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http://dx.doi.org/10.5604/01.3001.0014.1614DOI Listing
June 2020

Questionnaire survey on nurses and speech therapists regarding dysphagia rehabilitation in Japan.

Auris Nasus Larynx 2021 Apr 25;48(2):241-247. Epub 2020 Aug 25.

Department of Otolaryngology and Head and Neck Surgery, Tohoku University Graduate School of Medicine, 1-1 Seiryo-machi, Aoba-ku, Sendai 980-8574, Japan.

Objective: Current interventions of dysphagia are not generalizable, and treatments are commonly used in combination. We conducted a questionnaire survey on nurses and speech therapists regarding dysphagia rehabilitation to understand the current situation in Japan.

Methods: The questionnaire was sent to 616 certified nurses in dysphasia nursing and 254 certified speech-language-hearing therapists for dysphagia. Based on "Summaries of training methods in 2014" by JSDR, 24 local indirect exercises, 11 general indirect exercises, and 13 direct exercises were selected. The Likert scale "How do you feel about each method" was used as follows: A; Frequency, B; Ease, C; Adherence, D; Effectiveness (1-5))?".

Results: Two hundred fifty (40%) nurses and 145 (57%) speech-language-hearing therapists (ST) responded to the questionnaire. The direct exercise was associated with a significantly high score in every question. In indirect exercises, "Cervical range of motion exercise," "Orofacial myofunctional exercise," "Lip closure exercise." "Ice massage of pharynx" and "Huffing" were used relatively frequently. "Balloon dilatation therapy" and "Tube exercise" was associated with a relatively high discrepancy for two questions. Frequency" and the sum of "Ease," "Adherence," and "Effectiveness." was significantly correlated for local indirect exercises (r2 = 0.928, P < 0.01), general indirect exercises (r2 = 0.987, P < 0.01), and direct exercises (r2 = 0.996, P < 0.01) (Fig. 5).

Conclusion: This study examined the current situation of dysphagia rehabilitation in Japan. Our results aid to increase understanding and selection of rehabilitative treatments for dysphagia patients in Japan.
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http://dx.doi.org/10.1016/j.anl.2020.08.004DOI Listing
April 2021

Pulsatile tinnitus caused by pneumocephalus after Janneta surgery.

Auris Nasus Larynx 2021 Aug 22;48(4):793-796. Epub 2020 Jun 22.

Sen-En Rifu Otological Surgery Center, Miyagi, Japan.

Pulsatile tinnitus of nonvascular origin is rare. We herein present a case of pulsatile tinnitus complicated with Jannetta surgery due to a communication created between the drilled mastoid cells and epidural space. She was successfully cured by otological surgery where the mastoid tip was packed with bone cement. A 68-year-old woman was referred to the previous hospital with complaints of right autophony, aural fullness, hyperacusis to her footsteps, and pulsatile tinnitus for the past three years. She had received Jannetta surgery for right hemifacial spasm seven years before. The computed tomography (CT) of the right temporal bone showed bony dehiscence between the mastoid cells and posterior cranial fossa. She underwent otological surgery to obliterate the tip of the mastoid cavity with artificial bone cement (BIOPEX) under general anesthesia. Her annoying aural symptoms were immediately abolished and she has been free from symptoms at ten months after surgery. It is critical to ensure the closure of any communication created between the middle ear and epidural space during surgeries in order to prevent the occurrence of pulsatile tinnitus.
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http://dx.doi.org/10.1016/j.anl.2020.05.024DOI Listing
August 2021

Sialodochitis fibrinosa: Salivary duct obstruction by eosinophil extracellular traps?

Oral Dis 2020 Oct 11;26(7):1459-1463. Epub 2020 Jun 11.

Department of General Internal Medicine and Clinical Laboratory Medicine, Akita University Graduate School of Medicine, Akita, Japan.

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http://dx.doi.org/10.1111/odi.13434DOI Listing
October 2020

Diagnosis of the Patulous Eustachian Tube.

Ear Nose Throat J 2020 May 13:145561320925938. Epub 2020 May 13.

Sen-En Rifu Otological Surgery Center, Miyagi, Japan.

Objectives: To review the diagnosis of patulous Eustachian tube (PET) based on the diagnostic criteria for the PET proposed by Japan Otological Society (JOS).

Methods: We reviewed typical aural symptoms of PET, Eustachian tube (ET) obstruction procedure to confirm diagnosis of PET, objective findings of a patent ET obtainable from observation of the movement of the tympanic membrane, and by ET function tests (tubo-tympano-aerodynamic graphy, sonotubometry). In addition, usefulness of other tests such as patulous Eustachian tube handicap inventory-10 (PHI-10), sonotubometry with postural change (Ohta method), and sitting computed tomography (CT) to diagnose PET is described.

Results And Conclusions: We have described the diagnosis of PET based on the diagnostic criteria for PET proposed by JOS; PHI-10, Ohta method, and sitting CT are also useful for the diagnosis of PET. Further investigation is needed for an accurate diagnosis and precise evaluation of the pathophysiology of this challenging disease.
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http://dx.doi.org/10.1177/0145561320925938DOI Listing
May 2020

Treatment of oral ranula in HIV-positive patient.

Auris Nasus Larynx 2021 Feb 5;48(1):171-174. Epub 2020 Apr 5.

Division of Otolaryngology, Tohoku Medical and Pharmaceutical University, Sendai 981-0905, Japan. Electronic address:

HIV-associated salivary gland disease refers to the pathology in head and neck lesions such as ranula, salivary gland swelling, xerostomia, and benign lymphoepithelial cysts in the parotid gland. Here, we present a unique case of the ranula patient with HIV infection treated with OK-423 sclerotherapy. Case report: The patient was a 42-year-old Japanese male with a few months history of oral floor swelling. Computed tomography (CT) showed a low-density area limited within the right floor of the mouth. Magnetic resonance imaging (MRI) revealed a distinct T2-high intensity area localized on the same location. The puncture fluid was bloody mucus, and the cytology was no malignancy. We diagnosed a simple ranula. He was, however, found to be HIV-antibody positive at the examination before treatment by chance. He was referred to the department of infectious diseases and definitively diagnosed HIV infection by western blot. We chose OK-432 sclerotherapy because of its minimally invasive and the risk of HIV infecting medical staff. Two times OK-432 injection made the lesion disappear. Conclusion: The case indicated that OK-432 sclerotherapy could be effective for ranula related to HIV.
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http://dx.doi.org/10.1016/j.anl.2020.02.009DOI Listing
February 2021

Location of the stapedius muscle with reference to the facial nerve in patients with unilateral congenital aural atresia: implication for active middle ear implants surgery.

Acta Otolaryngol 2020 Jun 18;140(6):445-449. Epub 2020 Feb 18.

Sen-En Rifu Otologic Surgery Center, Miyagi, Japan.

Detailed investigations of the stapedial muscle (SM) in congenital aural atresia (CAA) patients have yet to be adequately conducted. To assess image variations in the mastoid segment of the facial nerve (FN) and SM in CAA. A total of nine patients comprising of 9 ears with unilateral CAA were studied. The courses of the FN and SM were evaluated from the basic point to 1 mm intervals between the mastoid portion of FN, and measured from the mean X and Y values in each group. The atresia side of FN among the Y values showed significant differences compared to the contralateral side. In terms of the SM, there were no significant differences in both the X and Y values. The stapedial muscle of the CAA patients was located medially to the FN. Conversely, the distance from the PSC to the FN revealed no significant differences with regard to the X and Y values for each group. The current observations revealed that the SM is located more posterior to the FN in CAA patients, and this is mainly attributed to the laterally and anteriorly displaced FN.
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http://dx.doi.org/10.1080/00016489.2020.1725113DOI Listing
June 2020

The characteristic of patulous eustachian tube patients diagnosed by the JOS diagnostic criteria.

PLoS One 2019 27;14(12):e0226908. Epub 2019 Dec 27.

Department of Otolaryngology-Head and Neck Surgery, Tohoku University, Graduate School of Medicine, Sendai, Miyagi, Japan.

Objective: The objective is to describe characteristics of patients diagnosed with patulous Eustachian tube (PET) using the Diagnostic Criteria proposed by Japan Otological Society, and to evaluate the efficiency of objective tests to determine patent Eustachian tube.

Study Design: Retrospective.

Setting: Tertiary referral center.

Subjects: A retrospective survey of medical records in Sen-En Rifu Hospital identified 78 ears of 56 patients with "Definite PET" diagnosed by the JOS Diagnostic Criteria between January 2017 and December 2017.

Method: Initial diagnosis, aural symptoms (voice autophony, aural fullness and breathing autophony), tubal obstruction procedures (posture change and pharyngeal orifice obstruction) and objective findings (tympanic membrane movement, Tubo-Tympano-Aerodynamic Graphy (TTAG) and sonotubometry) were evaluated. In addition, sonotubometry with postural change (Ohta's method), sitting CT and a newly devised PHI-10 score were also examined.

Results: Voice autophony, aural fullness, and breathing autophony were observed in 93.6%, 87.2%, 78.2%, respectively. In 91% of the ears, PET symptoms improved by postural change from sitting to the lying / forward-bending position. Synchronous movement of the TM upon respiration was observed in 69.1% of the ears. Positive findings of TTAG were observed in 75.6% of ears. Positive findings of sonotubometry were found in 55.1% of ears. Sonotubometry with postural change (Ohta's method), when the cut-off value of over 10dB was used, was positive in 45.2% of ears. Newly devised PHI-10 score representing severity of subjective symptoms classifying patients into no handicap, mild handicap, moderate handicap and severe handicap were observed in 12.2%, 10.8%, 18.9% and 58.1% of ears, respectively. The evaluation of the extent of patency of the ET by sitting CT indicated completely open, closed-short, and closed-long, in 68.6%, 11.4% and 21.4% of ears, respectively. Compared to the closed group, the completely open group had a significantly higher frequency of positive breathing autophony, positive sonotubometry, and positive Ohta's method.

Conclusion: The characteristics of main symptoms and the efficiency of various tests in PET diagnosis were analyzed based on data obtained from "Definite PET" patients diagnosed by the JOS Diagnostic Criteria. The greater the availability of tests to evaluate PET, the greater the opportunities to diagnose "Definite PET". In particular, tests measuring pressure transmission between the nasopharynx and middle ear, such as TM observation and TTAG, are more sensitive than sonotubometry measuring sound transmission.
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http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0226908PLOS
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6934284PMC
April 2020

Malignancies in Patients with IgG4-Related Diseases in Head and Neck Regions.

Tohoku J Exp Med 2019 12;249(4):285-290

Division of Gastroenterology, Tohoku Medical and Pharmaceutical University.

Immunoglobulin G4-related disease (IgG4-RD) is a recently recognized disease, characterized by high serum IgG4 concentrations and IgG4-producing plasma cell expansion with fibrotic or sclerotic changes in affected organs. Recent work has focused on the relationship between IgG4-RD and malignancies, but there is no report of malignancies associated with IgG4-RD in head and neck regions. The aim of this study was to analyze the clinicopathological characteristics of malignancies in patients with IgG4-RD in head and neck regions. We retrospectively analyzed 26 patients with IgG4-RD (12 men and 14 women aged 60.6 ± 11.6 years). The mean follow-up period was 26.6 months (from 12 to 96 months). These patients were divided into single-lesion group (n = 12) with IgG4-RD only in head and neck regions and multiple-lesion group (n = 14) with IgG4-RD in other regions. There was no significant difference in serum IgG4 concentrations between the single-lesion group (459.4 ± 336.4 mg/dL) and the multiple-lesion group (908.0 ± 739.2 mg/dL) (P = 0.07), whereas the IgG4/IgG ratio was significantly lower in the single-lesion group (22.8 ± 11.0%; n = 11) compared with the multiple-lesion group (31.7 ± 15.0%; n = 11, P = 0.02). Among the 26 patients, two patients (7.7%), both in the multiple-lesion group, developed life-threatening malignancies (salivary duct carcinoma in the submandibular gland and lymphoma in the orbital tissue). All physicians need to keep in mind the possible coexistence of malignancies in patients with IgG4-RD with high IgG4/IgG ratio and multiple lesions at the time of diagnosis.
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http://dx.doi.org/10.1620/tjem.249.285DOI Listing
December 2019

Hearing Recovery After Ejection of Air in a Case of Traumatic Pneumolabyrinth: Mechanism and Management Options.

Otol Neurotol 2020 03;41(3):359-363

Department of Otolaryngology, Jichi-Medical University.

Objective: To describe a case of traumatic pneumolabyrinth and subsequent hearing recovery after ejection of air with transcanal endoscopic surgical exploration.

Patients: A 38-year-old man was struck by his child while cleaning his ear with an ear pick made of bamboo, which penetrated deep into the left ear canal. Severe vertigo with vomiting and left hearing impairment ensued. In addition, high-resolution computed tomography demonstrated an air density within the vestibule.

Interventions: Exploratory tympanotomy was performed endoscopically a day after the injury and air was ejected from the oval window surgically.

Main Outcome Measures: High-resolution computed tomography, audiologic testing.

Results: Several hours after surgery, the patient's subjective vestibular symptoms lessened and 7 days after surgery, the patient felt slight dizziness when moving his head and no apparent spontaneous nystagmus was observed with an infrared charge-coupled device camera and was discharged from the hospital. Two years later, there are no subjective vestibular symptoms at all and the pure-tone average of his left ear improved to 16.7 dB.

Conclusion: We presented a case of traumatic pneumolabyrinth and the subsequent hearing recovery after ejection of air following endoscopic exploratory tympanotomy. We propose that initial management for traumatic pneumolabyrinth should be ejection of the air bubble if it is located solely in the vestibule and sparing the cochlea.
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http://dx.doi.org/10.1097/MAO.0000000000002538DOI Listing
March 2020

Nodal Merkel Cell Carcinoma in Head and Neck Lesions with an Unknown Primary: A Case Report in Light of the Literature.

Yonago Acta Med 2019 Sep 13;62(3):258-262. Epub 2019 Sep 13.

Division of Pathology, Tohoku Medical and Pharmaceutical University, Sendai 981-0905, Japan.

Merkel cell carcinoma (MCC) is a rare but aggressive neuroendocrine skin cancer. To diagnose nodal MCC with an unknown primary disease is challenging, and it has to be separated from other nodal metastatic neoplasms. We report a unique case of nodal MCC in head and neck lesions with an unknown primary. A 70-year-old woman was admitted to our department with a right submandibular mass. Fine needle aspiration biopsy was performed and indicated malignancy. F-18-fluorodeoxyglucose positron emission tomography (PET) demonstrated abnormal accumulation in the right submandibular lymph node, right palatine tonsil, and right thyroid gland. For diagnostics and treatment, bilateral selective neck lymph node dissection, right tonsillectomy, and right thyroidectomy were performed. Histopathological examination revealed that most parts of the submandibular lymph node were occupied by diffuse sheets of tumor cells. Contrary to our expectation, malignant cells were not detected in the right palatine tonsil and right thyroid. Immunohistochemistry demonstrated a marked positive reaction for AE1/AE3, chromogranin A, synaptophysin, cytokeratin 20 (CK20) and CD56 and a negative reaction for vimentin, leucocyte common antigen (LCA), thyroid transcription factor-1 (TTF1) and cytokeratin 7 (CK7) in the tumor cells. Immunostaining of Merkel cell polyomavirus-large T antigen (MCPyV-LT) showed a positive reaction and MCPyV-positive MCCs were assessed by PCR analysis, demonstrating that viral copy number was 12.8 copies per cell. These histological findings confirmed the diagnosis of Merkel cell carcinoma of the lymph node. In cases of tumors in the lymph node with a neuroendocrine appearance in head and neck lesions, it is necessary to eliminate the possibility of metastasis from MCC.
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http://dx.doi.org/10.33160/yam.2019.09.003DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6739247PMC
September 2019

Plug size selection protocol for the treatment of intractable patulous Eustachian tube with Kobayashi Plug.

Acta Otolaryngol 2019 Oct 20;139(10):849-853. Epub 2019 Aug 20.

Department of Otolaryngology-Head and Neck Surgery, Tohoku University, Graduate School of Medicine , Sendai , Japan.

: There have been no useful criteria for initial plug size selection protocol for the treatment of intractable patulous Eustachian tube (PET). : To establish a method for appropriate plug size selection using tubal function test and subjective symptom severity in PET patients who were treated by Kobayashi Plug insertion. : A retrospective survey of medical records identified 39 ears of 35 patients with PET who received insertion of the Kobayashi Plug and whose PET symptoms were thereafter controlled for at least 6 months after surgery. : The evaluation scale of PET handicap inventory-10 (PHI-10) was used to indicate PET subjective symptom severity. Tubal function tests (sonotubometry and tubo-tympano-aerodynamic-graphy: TTAG) were performed. : There was no correlation between the preoperative PHI 10 score and plug size ( = .157). There was a significant correlation between the preoperative sound attenuation from nostril to EAC measured by sonotubometry and plug size ( < .001). There was no correlation between the preoperative pressure transmission ratio estimated by TTAG and plug size ( = .271). Sonotubometry which evaluates sound attenuation from nostril to EAC can be a useful tool for selecting plug size.
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http://dx.doi.org/10.1080/00016489.2019.1651939DOI Listing
October 2019

Efficacy of a silicone plug for patulous eustachian tube: A prospective, multicenter case series.

Laryngoscope 2020 05 10;130(5):1304-1309. Epub 2019 Aug 10.

Department of Otolaryngology-Head and Neck Surgery, Tohoku University Graduate School of Medicine, Sendai, Japan.

Objectives/hypothesis: To report the efficacy and safety of transtympanic plugging of the eustachian tube (ET) using a silicone plug (Kobayashi plug) for chronic patulous ET (PET).

Study Design: Prospective and multicenter trial conducted in which 30 PET patients were resistant to at least 6 months of conservative treatment.

Methods: The efficacy and safety of 28 and 27 patients, respectively, were analyzed. All patients fulfilled inclusion and exclusion criteria. The primary end point used the patulous eustachian tube handicap inventory-10 (PHI-10), and the secondary end point used ET function tests such as sonotubometry, tubo-tympano-aerodynamic-graphy, and respiratory movement of the tympanic membrane and auscultation of voice sounds transmitted from the nose through the ET to the external auditory canal at 3months after surgery.

Results: PHI-10 scores were 34.4 ± 4.2, 6.4 ± 9, and 5.7 ± 8.6 at screening, and 3 and 6 months after surgery. Twenty-three cases (82.1%, 95% confidence interval: 63.1%-93.9%) were judged as successes. There were five cases (17.2%) of middle ear effusion, four cases (13.8%) of tympanic membrane perforation, and one case of tinnitus due to surgery to remove the plug. No severe or life-threatening complications were found.

Conclusions: This study revealed the efficacy and safety of silicone plug insertion for severe PET patients.

Level Of Evidence: 2 Laryngoscope, 130:1304-1309, 2020.
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http://dx.doi.org/10.1002/lary.28229DOI Listing
May 2020

Treatment of Auricular Hematomas by OK-432: How and Why It Works.

Otol Neurotol 2019 09;40(8):e820-e823

Division of Medical Biochemistry, Department of Biomolecular Sciences, Saga Medical School, Saga, Japan.

Objectives: The aim of this article was to investigate the effectiveness and underlying mechanisms of OK-432 therapy in patients with auricular hematomas.

Study Design And Setting: Case series with planned data collection.

Subjects And Methods: We tried this therapy in 47 patients with auricular hematoma between April 2008 and August 2018. We aspirated as much of the fluid content of each lesion as possible with a 21-gage needle. We injected OK-432 solution into the lesion with the same needle that we used for aspiration. We performed this treatment at an outpatient basis without hospitalization.

Results: Disappearance and marked reduction of the lesion were observed in all patients who had this therapy, and local scarring and deformity of the auricle did not occur in any patients. As adverse effects, local pain at the injection site and fever (37-38.5°C) were observed in some cases of the patients who had this therapy. The concentrations of various cytokines in each aspirate before and after OK-432 therapy were investigated. The production of tumor necrosis factor-α, interleukin-6, interleukin-8, interferon gamma, vascular endothelial growth factor, and periostin was significantly elevated in the aspirate fluid after OK-432 therapy.

Conclusion: OK-432 therapy is simple, easy, safe, effective, and can be used as a substitute for surgery in the treatment of auricular hematoma.
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http://dx.doi.org/10.1097/MAO.0000000000002336DOI Listing
September 2019

Risk factors of post-tonsillectomy dysgeusia.

Auris Nasus Larynx 2020 Apr 16;47(2):238-241. Epub 2019 Jul 16.

Department of Otolaryngology-Head and Neck Surgery, Tohoku University Graduate School of Medicine, Sendai, Japan. Electronic address:

Objective: To investigate risk factors for dysgeusia after tonsillectomy.

Methods: A retrospective survey of medical records identified 318 patients (163 male and 155 female subjects aged 12-75 years, average 41.7±16.1 years) who underwent tonsillectomy.

Results: Thirty three patients (10.4%) and seven patients (2.2%) reported suffering from dysgeusia three and six months after tonsillectomy, respectively. Average age, serum level of Zn and operation time were not significantly different between positive and negative findings of dysgeusia. Women with positive findings of dysgeusia (72.7%) was significantly higher than men (27.3%) (Odds ratio: 3.276, 95% CI: 1.441-7.450, p<0.005). A chi-square test of independence was calculated comparing the frequency of dysgeusia in under 60 years old group (13-59 years-old) and 60 years old and over group (60-75 years-old). A significant interaction was found (χ2 (1)=5.238. p<0.05).

Conclusions: Women and under 60 years old patients had a significantly higher rate of dysgeusia after tonsillectomy. It is recommended that patients are informed of the potential postoperative complication of dysgeusia.
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http://dx.doi.org/10.1016/j.anl.2019.07.005DOI Listing
April 2020

Endoscopic Treatment of Sinonasal Glomangiopericytoma: A Case Report in Light of the Literature.

Yonago Acta Med 2019 Jun 20;62(2):236-239. Epub 2019 Jun 20.

Division of Pathology, Tohoku Medical and Pharmaceutical University, Sendai 981-0905, Japan.

A 71-year-old Japanese male patient presented with a rare case of Glomangiopericytoma (GPC) of the left nasal with obstruction. Complete resection with endoscopic surgery was performed. Immunohistochemical staining for smooth muscle actin, β catenin, cyclin D1, vimentin, and factor 13 were helpful in establishing a definitive diagnosis. Extranasal treatment has been traditionally performed for successful management. However, recent advances in endoscopic treatment have enabled complete endoscopic resection of GPC, minimizing morbidity and facilitating subsequent surveillance for recurrence. Endoscopic management should be considered in suitable cases.
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http://dx.doi.org/10.33160/yam.2019.06.009DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6584261PMC
June 2019

The expression of 11 beta hydroxysteroid dehydrogenase in severe allergic rhinitis.

Otolaryngol Pol 2018 Oct;73(2):18-22

Tohoku Medical and Pharmaceutical University Otolaryngology.

Objective: To clarify the roles of 11 beta-HSD in resistance to glucocorticoid therapy for allergic rhinitis, a case series study was conducted.

Methods: The patient group consisted of 20 subjects with allergic rhinitis, aged from 21 to 46 years (mean age 26.5), who showed persistent GC resistance necessitating surgical removal of the inferior turbinate after 6 months' GC treatment. The patients with poor response to GC treatment for 6 months' were defined as GC resistance. The control group consisted of 10 subjects aged from 16 to 39 years (mean age 24.5) who underwent maxillofacial surgery, from whom nasal tissues were taken and who did not receive GC treatment. Nasal mucosal tissues from patients and cntorol subjects were examined immunohistochemically. The sections were washed with 0.01 M phosphate-buffered saline (PBS; pH 7.2) containing 0.15 M NaCl and 0.01% Triton X-100, and incubated for 2 h with rabbit polyclonal anti-11 beta HSD1 and 11 beta-HSD2 antibody (Santa Cruz Biotechnology, Inc., Santa Cruz, CA, USA), each diluted 1:200 in PBS containing 0.1% bovine serum albumin. Immunostained sections were assessed under an Olympus microscope with an eyepiece reticule at 200 X magnification. Cell counts are expressed as means per high-power field (0.202 mm2). Control group means (arithmetic mean ± SD) were compared with patient group means by Mann-Whitney U-test at P = 0.05.

Results: Although 11 beta-HSD1 was expressed to a similar extent in patients and controls, 11 beta-HSD2 was expressed significantly more in patients with severe allergic rhinitis, resulting in a increased HSD-1/HSD-2 ratio. The significantly increased expression of 11 beta-HSD2 in the nasal epithelium and submucosal inflammatory cells of patients with severe nasal allergy were observed in the present study.

Conclusion: Our findings suggest that 11 beta-HSD2 plays an important role in resistance to glucocorticoid therapy for allergic rhinitis, and its expression might be used as an additional parameter indicating steroid resistance in allergic rhinitis.
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http://dx.doi.org/10.5604/01.3001.0012.6143DOI Listing
October 2018

Width of Patulous Eustachian Tube: Comparison of Assessment by Sonotubometry and Tubo-tympano-aerography.

Otol Neurotol 2019 04;40(4):e386-e392

Sen-En Rifu Otologic Surgery Center, Miyagi-gun.

Objective: To assess the accuracy of tubo-tympano-aerodynamic-graphy (TTAG) in the estimation of morphological severity of patulous Eustachian tube (PET), and to compare the results with that of sonotubometry in clinical cases and in model experiments.

Study Design: Retrospective chart review and model experiment.

Setting: Tertiary referral center.

Subjects: A retrospective survey of medical records in Sen-En Rifu Hospital identified 26 patients (12 men and 14 women subjects aged 14-78 yr, average 49.4 ± 20.9 yr), comprising of 35 ears with PET. A definite diagnosis of PET was made for all patients based on the diagnostic criteria proposed by the Japan Otological Society.

Main Outcome Measures: Commercial equipment for sonotubometry and TTAG (JK-05A; RION Co., Ltd., Kokubunji, Tokyo, Japan) were used in the following investigations. Model Experiment: The ET model was made using two truncated syringes with silicone barrels and a narrow connecting tube (0.2, 0.4, 0.58, 0.8, 1.00, 2.00, and 3.00 mm). Clinical Investigation: Morphological evaluation of ET patency was performed by a three-dimensional (3-D) CT (3D Accuitomo; Morita MFG. Corp., Kyoto, Japan) and analyzed by software (Integrated Information System i-VIEW; Morita MFG. Corp.).

Results: The transmitted sound pressure level gradually increased as the tube caliber was widened up to 3 mm diameter. On the other hand, pressure transmission was suddenly elevated and reached a plateau at 0.58 mm in diameter. The cross-sectional area at the narrowest portion of the ET was significantly correlated with the sound attenuation from nostril to external auditory canal (EAC) (Spearman rank correlation test: r = -0.521, p < 0.001). In contrast, the cross-sectional area at the narrowest portion of the ET was not correlated with the pressure changes of TTAG (Spearman rank correlation test: r = 0.317, p = 0.64).

Conclusion: Sound transmission assessed by sonotubometry could be more useful than pressure transmission assessed by TTAG to predict the morphological severity of PET.
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http://dx.doi.org/10.1097/MAO.0000000000002141DOI Listing
April 2019

Surgical treatments for a case of superior canal dehiscence syndrome associated with patulous Eustachian tube.

Auris Nasus Larynx 2019 Aug 2;46(4):630-635. Epub 2018 Oct 2.

Sen-En Rifu Otological Surgery Center, Miyagi, Japan. Electronic address:

Objectives: The patulous Eustachian tube (PET) and superior semicircular canal dehiscence syndrome (SCDS) have similarity in their symptoms and similar effects caused by positional changes, causing difficulty in the differentiation between the two disorders. This report describes a case of both SCDS and PET that was eventually successfully treated.

Methods: A 68-year-old man presented with hyperacusis to his own footsteps and gait disturbance. He had been diagnosed as PET two years before and had been treated by insertion of a silicone plug (Kobayashi plug) at the other hospital. Clinical case records, audiological data, cervical vestibular-evoked myogenic potential (cVEMP), Eustachian tube function tests and computed tomography (CT) were taken in the sitting position.

Results: While the CT confirmed superior semicircular canal dehiscence, the results of cVEMP was not typical of SCD likely due to preexisting hearing impairment in the right ear with a history of middle ear surgeries for the treatment of PET. He received round window reinforcement (RWR) and achieved relief from his symptoms but six months after the surgery, he visited again with complaints of autophony of his own voice and breathing. The tympanic membrane was found to move synchronous with respiration, and Eustachian tube function tests and the sitting CT confirmed the recurrence of severe PET. He had his silicone plug exchanged (increase in size of the Kobayashi plug) and achieved relief from symptoms.

Conclusions: The present case was a rare instance showing that PET and SCDS can occur simultaneously in a patient. The patient achieved relief from symptoms after treatment with RWR and insertion of the Kobayashi plug.
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http://dx.doi.org/10.1016/j.anl.2018.09.008DOI Listing
August 2019

Incidence of Functional Nasal Voice in Patients With Patulous Eustachian Tube.

Otol Neurotol 2018 12;39(10):e1034-e1038

Sen-En Rifu Otologic Surgery Center.

Objective: Some patients with a patulous Eustachian tube (PET) complain of a nasal voice. This feature is often dismissed without further investigation. As such, there are only a few reports on this important symptom and scant studies have been conducted on a sufficiently large number of cases with PET. Therefore, this study was undertaken to investigate the characteristics of patients having a nasal voice and to examine whether this symptom can be an indication of the severity of PET.

Study Design: Retrospective.

Setting: Tertiary referral center.

Subjects And Methods: A retrospective survey of medical records in Sen-En Rifu Hospital identified 85 patients (40 men and 45 women) with PET between 2013 and 2016. Diagnosis of definite PET was based on the Proposal on Diagnostic Criteria of PET announced by the Otological Society of Japan (2017). The questionnaire inquired about the presence of a nasal voice and it was distributed to each patient at the first visit to the clinic. If a patient marked "yes" for the presence of nasal voice, he/she was later asked on the telephone to exclude nasal voice ascribable to causes other than PET, such as nasal diseases. Correlation between nasal voice and patient characteristics (age, sex, affected side, and PET symptoms such as autophony of own voice, aural fullness, and autophony of breathing sounds), subjective severity of PET evaluated by patulous Eustachian tube handicap inventory-10 (PHI-10), and that with the objective severity of PET evaluated by tubo-tympano-aerodynamic-graphy (TTAG) and sonotubometry were investigated.

Results: Seventy-six patients (36 men and 40 women) with definite PET were evaluated in this study. Thirteen patients (17.1%) (five men and eight women) reported a nasal voice coinciding with the occurrence of PET symptoms such as voice autophony, aural fullness, and breathing autophony. Age, sex, affected side, PET symptoms (autophony of their own voice, aural fullness, and autophony of their breathing sounds), and objective findings (TTAG and sonotubometry) were not significantly different between the two groups. The average total score of the PHI-10 in the "PET associated Nasal Voice Group" was 35.8 ± 4.5, which was statistically higher than that of the "non PET associated Nasal Voice Group" 23.6 ± 10.7 (p = 0.002). Out of 76 patients, 44 were treated surgically (Kobayashi Plug). In the "PET associated Nasal Voice Group," 85% (11 out of 13) were subjected to surgical treatment, whereas 52% (33 out of 63) underwent surgical treatment in the "non PET associated Nasal Voice Group." The rate of surgical treatment was significantly higher in "PET associated Nasal Voice Group" (p = 0.047).

Conclusion: Nasal voice due to PET symptoms was observed in 17.1% of PET patients. It was generally found in patients with severe subjective symptoms. Nasal voice can be an indication of subjective severity. However, this study failed to show objective evidence of wider Eustachian tube in such cases. Patients with a nasal voice tended to seek vigorous treatment including surgery.
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http://dx.doi.org/10.1097/MAO.0000000000001981DOI Listing
December 2018

Vibrant Soundbridge implantation via a retrofacial approach in a patient with congenital aural atresia.

Auris Nasus Larynx 2019 Apr 7;46(2):204-209. Epub 2018 Sep 7.

Sen-En Rifu Otologic Surgery Center, Miyagi-gun, Miyagi, Japan.

Objective: A method of Vibrant Soundbridge (VSB) placement to the round window (RW) via the retrofacial approach with preoperative evaluation of the relationship between the facial nerve (FN) and RW by 3D-CT reconstruction was proposed for the treatment of congenital aural atresia (CAA) patient.

Methods: A fenestration to the mesotympanum was made mastoid portion of the FN. During the approach, part of the stapedial muscle was encountered and removed. The RW niche was identified, and the floating mass transducer was placed from an inferior approach into the RW niche.

Results: There were no intra-operative or post-operative surgical complications.

Conclusion: The VSB placement to the RW via the retrofacial approach with partial removal of the stapedial muscle can be feasible alternative in CAA cases associated with an anteriorly and laterally positioned aberrant FN. Preoperative assessment using 3D CT may facilitate in assessing the feasibility of the approach and implantation of VSB.
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http://dx.doi.org/10.1016/j.anl.2018.08.012DOI Listing
April 2019