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1.
Auris Nasus Larynx ; 51(4): 738-746, 2024 Jun 07.
Article in English | MEDLINE | ID: mdl-38850719

ABSTRACT

OBJECTIVE: The present study aimed to observe and analyze the ocular movements induced by Coriolis stimulation (eccentric pitch while rotating: PWR) that induces Coriolis forces on the vestibular apparatus of healthy human individuals. METHODS: A total of 31 healthy subjects participated in the study. Eccentric PWR was performed on 27 subjects, by pitching the participants' heads forward and backward at an angle of 30° each on an axis parallel and 7 cm below inter-aural axis, at a frequency of 0.5 Hz while on a chair rotating at a constant angular velocity of 97.2°/s on the earth-vertical axis. Ocular movements during stimulation were recorded using three-dimensional video-oculography. As a subsidiary analysis, 0.5 Hz head roll tilt was used as another stimulus that also induced torsional ocular movements. The forces induced on the vestibular apparatus, and phases of ocular torsion against the stimulus were calculated from the observed data. RESULTS: In the Coriolis stimulation during rightward yaw rotation, a rightward ocular torsion of 4.8° on average, was observed when the head pitched forward, and the direction of ocular torsion reversed when the head pitched backward. During leftward yaw rotation, these relationships were reversed with an average amplitude of 4.7° The phase of ocular torsion preceded that of Coriolis force by 0.2 s during rightward rotation and 0.14 s during leftward rotation. There were no significant differences in amplitude or phase between the directions of rotation. The phase lead of 0.5 Hz roll-tilt was significantly smaller than that of Coriolis stimulation (p < 0.01). CONCLUSION: Coriolis stimulation induced a specific pattern of ocular torsion, where its direction and phase suggested that the mechanism likely involved both the otolith and semicircular canals. Further studies may provide a clue to the magnitude of the otolith and semicircular canal contributions.

2.
Front Mol Neurosci ; 17: 1384764, 2024.
Article in English | MEDLINE | ID: mdl-38742227

ABSTRACT

Inner ear malformations are predominantly attributed to developmental arrest during the embryonic stage of membranous labyrinth development. Due to the inherent difficulty in clinically assessing the status of the membranous labyrinth, these malformations are diagnosed with radiographic imaging, based on the morphological characteristics of the bony labyrinth. While extensive research has elucidated the intricacies of membranous labyrinth development in mouse models, comprehensive investigations into the developmental trajectory of the bony labyrinth, especially about its calcification process, have been notably lacking. One of the most prominent types of inner ear malformations is known as incomplete partition (IP), characterized by nearly normal external cochlear appearance but pronounced irregularities in the morphology of the modiolus and inter-scalar septa. IP type II (IP-II), also known as Mondini dysplasia, is generally accompanied by an enlargement of the vestibular aqueduct and is primarily attributed to mutations in the SLC26A4 gene. In the case of IP-II, the modiolus and inter-scalar septa of the cochlear apex are underdeveloped or missing, resulting in the manifestation of a cystic structure on radiographic imaging. In this overview, we not only explore the normal development of the bony labyrinth in mice but also present our observations on otolith mineralization. Furthermore, we investigated the specifics of bony labyrinth and otolith mineralization in Slc26a4-deficient mice, which served as an animal model for IP-II. We ensured that these findings promise to provide valuable insights for the establishment of therapeutic interventions, optimal timing, targeted sites, and preventive measures when considering the management of this condition.

3.
Auris Nasus Larynx ; 51(3): 450-455, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38520976

ABSTRACT

OBJECTIVE: Short-term recurrence is common in patients with peritonsillar cellulitis and abscesses, leading to socioeconomic problems. Early switching from intravenous to oral antibiotics is feasible for treating certain diseases. However, reports on early switching and total antibiotic administration duration in peritonsillar cellulitis and abscesses are limited. This study aimed to determine the appropriate antibiotic therapy duration and examine the impact of early oral switch therapy on peritonsillar cellulitis and abscesses. METHODS: We retrospectively identified 98,394 patients who received antibiotic therapy during hospitalization for peritonsillar cellulitis and abscesses between July 1, 2010, and December 31, 2019, using the Japanese Diagnosis Procedure Combination database. RESULTS: Propensity score matching analysis revealed no significant between-group difference in the rehospitalization rate (early oral switch therapy and long intravenous therapy: 1.7 % [198 of 11,621] vs. 2.0 % [234 of 11,621], odds ratio [OR] 0.84, 95 % confidence interval [CI] 0.70-1.02). A long total duration of antibiotic therapy (reference: 1-9 days) was associated with a low risk of rehospitalization (10-14 days: OR 0.86, 95 % CI 0.78-0.95; 15+ days: OR 0.51, 95 % CI 0.38-0.66). CONCLUSION: Early oral switch therapy may be a viable option for treating patients with peritonsillar cellulitis and abscesses in good condition who can tolerate oral intake. No less than 10 days of antibiotic therapy is desirable.


Subject(s)
Anti-Bacterial Agents , Cellulitis , Peritonsillar Abscess , Recurrence , Humans , Female , Male , Anti-Bacterial Agents/therapeutic use , Anti-Bacterial Agents/administration & dosage , Retrospective Studies , Peritonsillar Abscess/drug therapy , Cellulitis/drug therapy , Middle Aged , Adult , Patient Readmission/statistics & numerical data , Aged , Administration, Oral , Propensity Score , Cohort Studies , Administration, Intravenous
4.
Auris Nasus Larynx ; 51(3): 525-530, 2024 Jun.
Article in English | MEDLINE | ID: mdl-38522357

ABSTRACT

OBJECTIVE: To evaluate the potential impact of coronavirus disease 2019 (COVID-19) and vaccinations on otologic diseases, including facial nerve paralysis (including Ramsay Hunt syndrome), vestibular neuritis, sudden sensorineural hearing loss, and Meniere's disease. METHODS: In this retrospective study, we conducted a time-series analysis employing a causal impact algorithm on a large-scale inpatient database in Japan. We compared the actual number of hospitalized patients with otologic diseases to two predictions: one without any covariates and another with a covariate accounting for the reduction in the number of hospitalized patients due to lockdown measures. Additionally, we performed Granger causality tests to ensure the robustness of our findings. RESULTS: No significant increase was noted in the number of hospitalized patients with otologic diseases following the onset of the COVID-19 pandemic in the causal impact analysis. Similarly, no notable surge was observed in hospitalizations for these diseases following the introduction of the COVID-19 vaccine. The Granger causality tests results aligned with the causal impact analysis findings. CONCLUSION: Our findings indicate that COVID-19 and vaccinations had minimal discernible effects on hospitalization of patients with otologic diseases, suggesting that otologic diseases may not be significantly impacted by COVID-19 and vaccinations, which could have implications for public health policies and the allocation of healthcare resources during a pandemic. Further research and monitoring of long-term effects are warranted to validate these findings and guide healthcare decision-making.


Subject(s)
COVID-19 , Hospitalization , Pandemics , SARS-CoV-2 , Humans , COVID-19/epidemiology , Japan/epidemiology , Retrospective Studies , Hospitalization/statistics & numerical data , Hospitalization/trends , Ear Diseases/epidemiology , COVID-19 Vaccines , Coronavirus Infections/epidemiology , Male , Pneumonia, Viral/epidemiology , Female , Betacoronavirus , Meniere Disease/epidemiology
5.
Acta Otolaryngol ; 144(2): 118-122, 2024 Feb.
Article in English | MEDLINE | ID: mdl-38546378

ABSTRACT

BACKGROUND: Laser fenestration in stapedotomy has thermal effect to the vestibule. AIMS/OBJECTIVES: To evaluate the role of energy density (fluence) in the severity of postoperative vestibular symptoms. MATERIALS AND METHODS: The retrospective chart-review study included 84 patients with otosclerosis that underwent primary laser stapedotomy. Surgical outcomes, including nystagmus, and subjective vestibular symptoms during one-month follow-up, were compared between potassium titanyl phosphate (KTP) and CO2 laser. According to this study and literature, we assessed the relationship between laser parameters and the incidence of persistent vestibular symptoms lasting more than one week after surgery. RESULTS: The KTP and CO2 laser group included 48 and 36 patients, respectively. Fluence was different between the KTP (637 J/cm2) and CO2 (141 J/cm2) laser (p < .001). The KTP group showed gradual decrease in dizziness during one-month observation period, while the CO2 group exhibited a steep recovery curve in the first postoperative week (9 and 4 d of duration, respectively). The incidence of persistent vestibular symptoms was correlated with both fluence (r = 0.80, p = .01) and spot size (r = -0.74, p = .01). CONCLUSIONS AND SIGNIFICANCE: Appropriate setting of parameters with lower fluence is desirable for the efficiency and safety of laser stapedotomy.Abbreviations: ABG: air-bone gap; SD: standard deviation.


Subject(s)
Dizziness , Lasers, Gas , Otosclerosis , Stapes Surgery , Humans , Stapes Surgery/methods , Stapes Surgery/adverse effects , Male , Female , Retrospective Studies , Middle Aged , Otosclerosis/surgery , Adult , Dizziness/etiology , Lasers, Gas/therapeutic use , Lasers, Solid-State/therapeutic use , Postoperative Complications/etiology , Postoperative Complications/epidemiology , Laser Therapy/adverse effects , Laser Therapy/methods , Aged
6.
Acta Otolaryngol ; 143(10): 849-855, 2023 Oct.
Article in English | MEDLINE | ID: mdl-38088257

ABSTRACT

BACKGROUND: Vestibular stimulation causes postural unsteadiness accompanied by a sensation of tilt. AIMS/OBJECTIVES: The mechanism of the sensation of tilt needs to be assessed by accurate calculation of the rotational axis of torsional eye position under various vestibular stimulations. MATERIAL AND METHODS: Twenty-two healthy subjects participated in the study. Thirteen subjects underwent bilateral vestibular stimulation by on-axis yaw rotation under various head positions, and eighteen subjects underwent unilateral vestibular stimulation by caloric irrigation under various head positions. Listing's Plane was plotted for the eye movement data obtained by three-dimensional video-oculography. RESULTS: The offset of Listing's Plane showed sustained deviation of torsional eye position that was more prominent in head positions that stimulated lateral semicircular canals more than vertical semicircular canals. There was a less prominent and directionally reversed offset in head positions that stimulated vertical canals more than lateral semicircular canals. CONCLUSION AND SIGNIFICANCE: The sustained torsional eye position was validated by accurate assessment using Listing's Plane. The mechanism behind the deviation may be due to a combination of multiple anatomical components within the vestibular apparatus, with potentially stronger influence from lateral semicircular canals.


Subject(s)
Reflex, Vestibulo-Ocular , Vestibule, Labyrinth , Humans , Reflex, Vestibulo-Ocular/physiology , Eye Movements , Semicircular Canals/physiology , Vestibule, Labyrinth/physiology , Rotation
7.
Hum Genet ; 142(10): 1499-1517, 2023 Oct.
Article in English | MEDLINE | ID: mdl-37668839

ABSTRACT

Enlargement of the endolymphatic sac, duct, and vestibular aqueduct (EVA) is the most common inner ear malformation identified in patients with sensorineural hearing loss. EVA is associated with pathogenic variants in SLC26A4. However, in European-Caucasian populations, about 50% of patients with EVA carry no pathogenic alleles of SLC26A4. We tested for the presence of variants in CHD7, a gene known to be associated with CHARGE syndrome, Kallmann syndrome, and hypogonadotropic hypogonadism, in a cohort of 34 families with EVA subjects without pathogenic alleles of SLC26A4. In two families, NM_017780.4: c.3553A > G [p.(Met1185Val)] and c.5390G > C [p.(Gly1797Ala)] were detected as monoallelic CHD7 variants in patients with EVA. At least one subject from each family had additional signs or potential signs of CHARGE syndrome but did not meet diagnostic criteria for CHARGE. In silico modeling of these two missense substitutions predicted detrimental effects upon CHD7 protein structure. Consistent with a role of CHD7 in this tissue, Chd7 transcript and protein were detected in all epithelial cells of the endolymphatic duct and sac of the developing mouse inner ear. These results suggest that some CHD7 variants can cause nonsyndromic hearing loss and EVA. CHD7 should be included in DNA sequence analyses to detect pathogenic variants in EVA patients. Chd7 expression and mutant phenotype data in mice suggest that CHD7 contributes to the formation or function of the endolymphatic sac and duct.


Subject(s)
Deafness , Hearing Loss, Sensorineural , Hearing Loss , Vestibular Aqueduct , Animals , Mice , Alleles , DNA Helicases/genetics , Hearing Loss/genetics , Hearing Loss, Sensorineural/genetics
8.
Laryngoscope Investig Otolaryngol ; 8(4): 1036-1043, 2023 Aug.
Article in English | MEDLINE | ID: mdl-37621296

ABSTRACT

Objective: We aimed to elucidate the ossification process of the otic capsule in postnatal C57BL/6 mice and depict the three-dimensional (3D) process of otoconial mineralization in vivo. Methods: The otic capsules of C57BL/6 mice were stained with alizarin red and imaged/compared using micro-computed tomography on postnatal day (P) between P0 and P8, P10, P15, and P30 and 3-4 months old (P3-4Mo). We reconstructed 3D images of the otic capsule and otoconia and measured the bone mineral density using x-ray absorptiometry on each age. Results: The 3D reconstructed otic capsule images revealed two ossification centers of the otic capsule at P0. One was observed around the ampulla of the superior semicircular canal and utricle, and the other was observed around the ampulla of the posterior semicircular canal. The cross-sectional views demonstrated that modiolar ossification developed from the base to the apex from P4 to P8. The inter-scalar septum ossified bidirectionally from the modiolus and bony otic capsule from P8 to P15. The mineralized otoconia were first detected in the utricle at P3 and saccular otoconia at P6. The density of the utricle and saccular otoconia showed different growth trends. Conclusion: To the best of our knowledge, this is the first study to demonstrate the 3D appearance of the otic capsule and otoconia in different developmental stages of mice. We also revealed that modiolar and inter-scalar septal calcification is the final event in the cochlea and that it can be susceptible to pathological conditions (cochlear congenital malformations and hereditary vestibular diseases). The unique features of the ossification process and duration may explain these pathological conditions observed in humans. Level of Evidence: 3.

9.
Otol Neurotol ; 44(7): 656-663, 2023 Aug 01.
Article in English | MEDLINE | ID: mdl-37400150

ABSTRACT

OBJECTIVE: To compare the findings of magnetic resonance imaging (MRI) with advanced protocols in patients with various types of acute sensorineural hearing loss (ASNHL). STUDY DESIGN: Retrospective case review. SETTING: Tertiary referral center. PATIENTS: Two hundred eighty-seven patients with ASNHL. INTERVENTIONS: All patients underwent MRI scanning, including heavily T2-weighted three-dimensional fluid-attenuated inversion recovery before and 4 hours after the intravenous administration of gadolinium contrast medium (delayed 3D-FLAIR). A hybrid of the reversed image of the positive endolymph signal and the native image of the perilymph signal image was constructed to visualize the endolymphatic space. RESULTS: The detection rates of abnormal MRI findings vary significantly among different types of ASNHL. A hyperintense signal on delayed 3D-FLAIR was observed in all patients with intralabyrinthine schwannoma or vestibular schwannoma and 20.5% of patients with idiopathic sudden sensorineural hearing loss (ISSNHL) but was rarely observed in definite Ménière's disease (MD, 2.6%). In contrast, endolymphatic hydrops (EH) was frequently observed in patients with definite MD (79.5%) but was observed much less frequently in patients with ISSNHL (11.0%). In patients with cochlear MD and ALHL, detection rates of cochlear EH were similar to those with definite MD, whereas detection rates of vestibular EH were significantly lower than in patients with definite MD. CONCLUSIONS: The significantly different detection rates of abnormal MRI findings among various types of ASNHL shed light on the distinct pathophysiology of each disorder. A diagnosis based on MRI findings with advanced protocols may help select treatment strategies and provide prognostic information for patients.


Subject(s)
Endolymphatic Hydrops , Hearing Loss, Sensorineural , Vestibule, Labyrinth , Humans , Retrospective Studies , Imaging, Three-Dimensional , Magnetic Resonance Imaging/methods , Hearing Loss, Sensorineural/diagnostic imaging , Contrast Media
10.
Neurobiol Dis ; 183: 106194, 2023 07.
Article in English | MEDLINE | ID: mdl-37295562

ABSTRACT

Patients with SLC26A4 mutations exhibit highly variable hearing loss and vestibular dysfunction. Although Slc26a4 mutant mice similarly exhibit vestibular deficits, including circling behavior, head tilting, and torticollis, the underlying pathogenesis of the vestibular symptoms remains unclear, hindering its effective management for patients with SLC26A4 mutations. In this study, we evaluated the equilibrium function using the inspection equipment, which can record eye movements against rotational, gravitational, and thermal stimulations. Moreover, we correlated the degree of functional impairment with the morphological alterations observed in Slc26a4Δ/Δ mice. The rotational stimulus and ice water caloric tests revealed considerable impairment of the semicircular canal, while the tilted gravitational stimulus test showed a severe functional decline of the otolithic system in Slc26a4Δ/Δ mice. Generally, the degree of impairment was more severe in circling Slc26a4Δ/Δ mice than in non-circling Slc26a4Δ/Δ mice. In non-circling Slc26a4Δ/Δ mice, the semicircular canal function was normal. Micro-computed tomography results showed enlargement of the vestibular aqueduct and bony semicircular canals but no correlative relationship between the severity of the caloric response and the size of bony labyrinths. Giant otoconia and a significant decrease in total otolith volume in the saccule and utricle were observed in Slc26a4Δ/Δ mice. However, the giant otoconia were not overly dislocated in the bony otolithic system and ectopic otoconia were absent in the semicircular canal. The number and morphology of the utricular hair cells in Slc26a4Δ/Δ mice were not significantly reduced compared to those in Slc26a4Δ/+ mice. Collectively, we can conclude that vestibular impairments are mainly associated with otoconia formation and morphology rather than hair cell degeneration. In addition, severe disturbances of semicircular canals cause circling behavior in Slc26a4Δ/Δ mice. Our comprehensive morphological and functional assessments apply to mouse models of other genetic diseases with vestibular impairment.


Subject(s)
Vestibular Aqueduct , Mice , Animals , X-Ray Microtomography , Sulfate Transporters/genetics , Mice, Knockout , Mutation
11.
Acta Otolaryngol ; 143(5): 387-391, 2023 May.
Article in English | MEDLINE | ID: mdl-37083024

ABSTRACT

BACKGROUND: Dynamic cervico- (COR) and vestibulo-ocular reflex (VOR) contribute to stabilise visual images in the retina. The gain in dynamic COR is small in healthy individuals but increases in patients with vestibular dysfunction. Conversely, static COR has not been directly observed in healthy individuals. OBJECTIVES: To elucidate the presence of static COR and quantify it in normal individuals in the roll plane. METHODS: Eleven healthy participants were included in the study. Eye torsions were measured using video oculography to evaluate the static COR induced by lateral neck flexion during a head-upright-with-body-tilt position at 15°, 30°, and 45°. The ocular counterroll (OCR) was compared during whole-body and head tilts to assess the influence of static COR on OCR. RESULTS: Static COR was significantly observed as eye torsion in the direction opposite to the body tilt. The head tilt OCR was significantly smaller than the whole-body tilt OCR to the right side but not to the left side. CONCLUSION: Static COR exists in healthy individuals and tends to show higher amplitude as neck flexion stimulation increases.


Subject(s)
Eye Movements , Reflex, Vestibulo-Ocular , Humans , Reflex, Vestibulo-Ocular/physiology , Posture/physiology , Neck , Face
12.
Laryngoscope Investig Otolaryngol ; 7(6): 2011-2019, 2022 Dec.
Article in English | MEDLINE | ID: mdl-36544934

ABSTRACT

Objective: To compare the microbiota between cholesteatoma and chronic suppurative otitis media (COM) and to identify potential pathogens that explain the relevant phenotypes of cholesteatoma. Study Design: Prospective cohort study. Methods: Surgical specimens collected from 20 cholesteatomas and nine COMs were treated to dissolve biofilms and subjected to 16S ribosomal RNA (rRNA) gene sequencing and amplicon sequence variant-level analysis for microbiota profiling and quantitative comparison. Correlations between the relative abundance of potential pathogens and the volume of the primary resected cholesteatomas were examined. Results: Differences in bacterial composition (beta diversity) were observed between cholesteatomas and COM (p = .002), with a higher abundance of Staphylococcus in cholesteatomas than in COM (p = .005). Common genera in the external auditory canal (EAC) flora, such as Staphylococcus, Corynebacterium, and Cutibacterium, were predominant in both cholesteatoma and COM; Staphylococcus aureus and Pseudomonas aeruginosa were increased in both diseases compared with the EAC flora. Furthermore, coagulase-negative staphylococci (CoNS) were more abundant in cholesteatomas than in COM (p = 0.002). Linear discriminant analysis coupled with effect size measurements (LEfSe) identified four CoNS as potential biomarkers for cholesteatoma. The relative abundance of S. aureus, a potential pathogen, was positively correlated with cholesteatoma volume (r = .60, p = .02). Conclusion: The microbiota of cholesteatoma and COM originated from EAC flora, but the bacterial composition was largely altered. Our results suggested that S. aureus infection is involved in cholesteatoma progression. Level of Evidence: 3b.

13.
Hum Genet ; 141(3-4): 455-464, 2022 Apr.
Article in English | MEDLINE | ID: mdl-34345941

ABSTRACT

Mutations of coding regions and splice sites of SLC26A4 cause Pendred syndrome and nonsyndromic recessive hearing loss DFNB4. SLC26A4 encodes pendrin, a transmembrane exchanger of anions and bases. The mutant SLC26A4 phenotype is characterized by inner ear malformations, including an enlarged vestibular aqueduct (EVA), incomplete cochlear partition type II and modiolar hypoplasia, progressive and fluctuating hearing loss, and vestibular dysfunction. A thyroid iodine organification defect can lead to multinodular goiter and distinguishes Pendred syndrome from DFNB4. Pendred syndrome and DFNB4 are each inherited as an autosomal recessive trait caused by biallelic mutations of SLC26A4 (M2). However, there are some EVA patients with only one detectable mutant allele (M1) of SLC26A4. In most European-Caucasian M1 patients, there is a haplotype that consists of 12 variants upstream of SLC26A4, called CEVA (Caucasian EVA), which acts as a pathogenic recessive allele in trans to mutations affecting the coding regions or splice sites of SLC26A4. This combination of an M1 genotype with the CEVA haplotype is associated with a less severe phenotype than the M2 genotype. The phenotype in EVA patients with no mutant alleles of SLC26A4 (M0) has a very low recurrence probability and is likely to be caused by other factors.


Subject(s)
Hearing Loss, Sensorineural , Hearing Loss , Deafness , Goiter, Nodular , Hearing Loss/genetics , Hearing Loss/pathology , Hearing Loss, Sensorineural/genetics , Hearing Loss, Sensorineural/pathology , Humans , Membrane Transport Proteins/genetics , Mutation , Phenotype , Sulfate Transporters/genetics , Vestibular Aqueduct/abnormalities
14.
J Assoc Res Otolaryngol ; 22(6): 681-691, 2021 12.
Article in English | MEDLINE | ID: mdl-34622375

ABSTRACT

Incomplete partition type II (IP-II) is frequently identified in ears with SLC26A4 mutations. Cochleae with IP-II are generally observed to have 1½ turns; the basal turns are normally formed, and the apical turn is dilated or cystic. The objective of this study was to characterize the pathomorphogenesis of the IP-II cochlear anomaly in Slc26a4-null mice. Otic capsules were dissected from Slc26a4Δ/+ and Slc26a4Δ/Δ mice at 1 and 8 days of age and at 1 and 3 months of age. X-ray micro-computed tomography was used to image samples. We used a multiplanar view and three-dimensional reconstructed models to calculate the cochlear duct length, cochlear turn rotation angle, and modiolus tilt angle. The number of inner hair cells was counted, and the length of the cochlear duct was measured in a whole-mount preparation of the membranous labyrinth. X-ray micro-computed tomography mid-modiolar planar views demonstrated cystic apical turns in Slc26a4Δ/Δ mice resulting from the loss or deossification of the interscalar septum, which morphologically resembles IP-II in humans. Planes vertical to the modiolus showed a similar mean rotation angle between Slc26a4Δ/+ and Slc26a4Δ/Δ mice. In contrast, the mean cochlear duct length and mean number of inner hair cells in Slc26a4Δ/Δ mice were significantly smaller than in Slc26a4Δ/+ mice. In addition, there were significant differences in the mean tilt angle and mean width of the modiolus. Our analysis of Slc26a4-null mice suggests that IP-II in humans reflects loss or deossification of the interscalar septum but not a decreased number of cochlear turns.


Subject(s)
Cochlea/abnormalities , Cochlea/diagnostic imaging , Ear, Inner/diagnostic imaging , Hair Cells, Vestibular , Sulfate Transporters/genetics , Animals , Cochlea/anatomy & histology , Cochlear Duct , Ear, Inner/abnormalities , Hair Cells, Auditory, Inner , Mice , Mice, Knockout , Tomography, X-Ray Computed , X-Ray Microtomography
16.
Auris Nasus Larynx ; 48(6): 1081-1091, 2021 Dec.
Article in English | MEDLINE | ID: mdl-34059399

ABSTRACT

OBJECTIVE: The recent development of extended reality technology has attracted interest in medicine. We explored the use of patient-specific virtual reality (VR) and mixed reality (MR) temporal bone models in anatomical teaching, pre-operative surgical planning and intra-operative surgical referencing. METHODS: VR and MR temporal bone models were created and visualized on head-mounted display (HMD) and MR headset respectively, by a novel webservice that allows users to convert computed tomography images to VR and MR images without specific knowledge of programming. Eleven otorhinolaryngology trainees and specialists were asked to manipulate the healthy VR temporal bone model and to assess its validity by filling out a questionnaire. Additionally, VR and MR pathological models of petrous apex cholesteatoma were utilized for surgical planning pre-operatively and for referring to the anatomy during the surgery. RESULTS: Most participants were favorable about the VR model and considered HMD as superior to a flat computer screen. 91% of the participants agreed or somewhat agreed that VR through HMD is cost effective. In addition, the VR pathological model was used for planning and sharing the surgical approach during a pre-operative surgical conference. The MR headset was worn intra-operatively to clarify the relationship between the pathological lesion and vital anatomical structures. CONCLUSION: Regardless of the participants' training level in otorhinolaryngology or VR experience, all participants agreed that the VR temporal bone model is useful for anatomical education. Furthermore, the creation of patient-specific VR and MR models using the webservice and their pre- and intra-operative usages indicated the potential of innovative adjunctive surgical instrument.


Subject(s)
Anatomy/education , Augmented Reality , Models, Anatomic , Otorhinolaryngologic Surgical Procedures/education , Temporal Bone/anatomy & histology , Virtual Reality , Humans , Intraoperative Care , Preoperative Care , Temporal Bone/diagnostic imaging , Temporal Bone/surgery , Tomography, X-Ray Computed
17.
J Vis Exp ; (169)2021 03 29.
Article in English | MEDLINE | ID: mdl-33843930

ABSTRACT

The study of mutant mouse models of human hearing and balance disorders has unraveled many structural and functional changes which may contribute to the human phenotypes. Although important progress has been done in the understanding of the development and function of the neurosensory epithelia of the cochlea and vestibula, limited knowledge is available regarding the development, cellular composition, molecular pathways and functional characteristics of the endolymphatic sac. This is, in large part, due to the difficulty of visualizing and microdissecting this tissue, which is an epithelium comprised of only one cell layer. The study presented here describes an approach to access and microdissect the endolymphatic sac from the wild-type mouse inner ear at different ages. The result of a similar dissection is shown in a pendrin-deficient mouse model of enlargement of the vestibular aqueduct. A transgenic mouse with a fluorescent endolymphatic sac is presented. This reporter mouse can be used to readily visualize the endolymphatic sac with limited dissection and determine its size. It can also be used as an educational tool to teach how to dissect the endolymphatic sac. These dissection procedures should facilitate further characterization of this understudied part of the inner ear.


Subject(s)
Endolymphatic Sac/surgery , Animals , Disease Models, Animal , Humans , Mice
18.
Front Neurol ; 11: 597562, 2020.
Article in English | MEDLINE | ID: mdl-33329351

ABSTRACT

The caloric step stimulus test consists of the changes in head position from the sitting to supine positions and continuous caloric irrigation. This test can provide a single labyrinth with a stimulus similar to constant head acceleration in rotational testing and, therefore, can evaluate vestibulo-ocular reflex (VOR) dynamics more precisely than can conventional methods. To assess the clinical utility of the test in the assessment of the VOR dynamics of diseases, we performed the test in patients with peripheral vestibular disorders, including sudden idiopathic hearing loss, vestibular neuritis, Meniere disease, vestibular Meniere disease, or chronic unilateral idiopathic vestibulopathy and normal controls. Slow-phase eye velocity (SPV) was measured with videonystagmography. We fitted the time course of SPV across 2 min to a mathematical model containing two exponential components and time constants: the caloric step VOR time constant (T 1) and caloric step VOR adaptation time constant (T 2). All responses of normal controls (n = 15 ears) were fit to the model. Several responses of the 101 ears of the patients differed from the time courses predicted by the model. We divided the data of 116 ears into four patterns based on SPV, T 1, and T 2. The thresholds for the classification were determined according to the lower limits of the capability of curve fitting for SPV and the upper limits of normal controls for T 1 and T 2. Seventy-eight ears followed pattern A (normal T 1 and T 2): the SPV trajectory formed a rapid rise with subsequent decay. Nineteen followed pattern B (normal T 1 and prolonged T 2): the SPV trajectory formed a rapid rise without decay. Six followed pattern C (prolonged T 1 and T 2): the SPV trajectory formed a slow rise. Thirteen ears followed pattern D: a low VOR response. There were no significant differences in time constants between the affected and healthy ears in patients with each disease. However, prolonged T 1 and T 2 were significantly more frequent in the affected ears than the healthy ears. In conclusion, the caloric step stimulus test can be potentially useful in detecting unusual VOR responses and thus reflect some pathological changes in the vestibular system.

19.
Laryngoscope Investig Otolaryngol ; 5(6): 1168-1175, 2020 Dec.
Article in English | MEDLINE | ID: mdl-33364409

ABSTRACT

OBJECTIVES: This study aimed to test the hypothesis that contrast-enhanced 3D MRI with gradient-echo sequences (CE-3D-GRE) can detect signs of vasculitis in the fallopian canal, which may cause otologic involvement, in four patients with antineutrophil cytoplasmic antibody-associated vasculitis (AAV). METHODS: CE-3D-GRE acquired at 3.0 Tesla was performed on four patients diagnosed with granulomatosis with polyangiitis or eosinophilic granulomatosis with polyangiitis, at onset or relapse of the disease, and in remission. Clinical correlations between otologic symptoms and radiological findings were examined for each patient. Furthermore, signal intensity of the mastoid segment of the fallopian canal was compared between the ears with active disease (n = 3) and those in remission or without vasculitis (n = 3). RESULTS: Intense enhancement in the tympanic and mastoid segments of the fallopian canal was associated with development of external otitis, otitis media, and sensorineural hearing loss, and was unrelated to the presence of facial paresis. Maximal intensity projection images visualized the close relationship between the enhanced fallopian canal and middle ear inflammation. The findings were absent in remission. Signal intensity of the mastoid segment of the fallopian canal was higher in ears with active disease than in normal ears (P < .001) and decreased to normal levels during remission (P = .597). CONCLUSION: CE-3D-GRE can demonstrate vasculitis in the temporal bone, reflecting disease activity and the severity of otologic manifestations, including cochlear involvement, in AAV patients. Intense enhancement of the fallopian canal on CE-3D-GRE can be a potential marker for vasculitis of the temporal bone. LEVEL OF EVIDENCE: 5.

20.
Acta Otolaryngol ; 140(11): 925-929, 2020 Nov.
Article in English | MEDLINE | ID: mdl-33026898

ABSTRACT

BACKGROUND: Management of vestibular falls requires precise evaluation of otolithic function and gravity recognition. Tilt of the head in the roll plane causes ocular counter rolling (OCR) with a gain of 5-10%, which can be a quantitative parameter related to the otolith-ocular reflex (OOR) in that plane. However, the OOR in the pitch plane is indiscernible because of our ability to voluntarily gaze and pursue. 3 D eye movements and its Listing's plane description can provide novel parameters with which to evaluate OOR, even in the pitch plane. OBJECTIVES: To evaluate the utility of 3 D eye movement analysis in video oculography (VOG) as a quantitative parameter reflecting otolithic function during head movements associated with daily life. MATERIALS AND METHODS: In healthy subjects, we analyzed the changes in 3 D eye movements and the Listing's plane that occur with static gravitational modification. RESULTS: OOR during head tilt in the roll plane revealed that OCR amplitude had a saturating/peaked conformation and was not fitted by a sine curve. OOR in the pitch plane revealed anteroposterior leaning of the Listing's plane. CONCLUSIONS: These novel tools for minimally invasive evaluation of OOR in a clinical setting could potentially enable us to quantitatively evaluate peripheral static disequilibrium.


Subject(s)
Eye Movement Measurements , Reflex, Vestibulo-Ocular , Vestibular Function Tests , Adult , Eye Movements , Female , Head/physiology , Humans , Male , Rotation , Vestibule, Labyrinth/physiology , Video Recording
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