Paula Robles-Bolivar, Marta Martínez-Martínez, Rocio Martín-Márquez, Inés Berrio-Domínguez, Jose Luis Martin-Rodríguez, Jose Antonio Lopez-Escamez
{"title":"未患梅尼埃病的人群中内淋巴囊发育不全的患病率","authors":"Paula Robles-Bolivar, Marta Martínez-Martínez, Rocio Martín-Márquez, Inés Berrio-Domínguez, Jose Luis Martin-Rodríguez, Jose Antonio Lopez-Escamez","doi":"10.1177/09574271251371541","DOIUrl":null,"url":null,"abstract":"<p><p>PurposeTo estimate the prevalence of endolymphatic sac hypoplasia (EShp)-a proposed specific finding in Ménière's disease (MD) that defines an endophenotype characterized by bilateral involvement, male predominance, temporal bone abnormalities, and familial clustering-in individuals without MD, to assess its specificity for the condition.MethodsWe analyzed 956 temporal bone CT scans from individuals without MD to assess the prevalence of EShp using the Angular Trajectory of the Vestibular Aqueduct (ATVA) marker. ATVA distribution, reproducibility, and associations with clinical variables were also evaluated.ResultsEShp was identified in 6 ears from 4 individuals, yielding a prevalence of 0.6% per patient and 0.8% per ear. ATVA values had a median of 95.0° (IQR = 12.5°, range 65.9°-159.4°). Interobserver agreement was good (ICC = 0.75), with a mean bias of 6.2° ± 5.4° and 5.5% of ears outside the 95% limits of agreement. No significant associations were found between ATVA and sex, age, or clinical diagnosis.ConclusionsEShp is rare in individuals without MD, supporting its specificity and potential role in MD pathophysiology. The ATVA marker is reliable and reproducible and may serve as a CT biomarker for the hypoplastic MD endophenotype.</p>","PeriodicalId":49960,"journal":{"name":"Journal of Vestibular Research-Equilibrium & Orientation","volume":" ","pages":"9574271251371541"},"PeriodicalIF":3.3000,"publicationDate":"2025-08-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Prevalence of the endolymphatic sac hypoplasia in a cohort of individuals without Meniere disease.\",\"authors\":\"Paula Robles-Bolivar, Marta Martínez-Martínez, Rocio Martín-Márquez, Inés Berrio-Domínguez, Jose Luis Martin-Rodríguez, Jose Antonio Lopez-Escamez\",\"doi\":\"10.1177/09574271251371541\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>PurposeTo estimate the prevalence of endolymphatic sac hypoplasia (EShp)-a proposed specific finding in Ménière's disease (MD) that defines an endophenotype characterized by bilateral involvement, male predominance, temporal bone abnormalities, and familial clustering-in individuals without MD, to assess its specificity for the condition.MethodsWe analyzed 956 temporal bone CT scans from individuals without MD to assess the prevalence of EShp using the Angular Trajectory of the Vestibular Aqueduct (ATVA) marker. ATVA distribution, reproducibility, and associations with clinical variables were also evaluated.ResultsEShp was identified in 6 ears from 4 individuals, yielding a prevalence of 0.6% per patient and 0.8% per ear. ATVA values had a median of 95.0° (IQR = 12.5°, range 65.9°-159.4°). Interobserver agreement was good (ICC = 0.75), with a mean bias of 6.2° ± 5.4° and 5.5% of ears outside the 95% limits of agreement. No significant associations were found between ATVA and sex, age, or clinical diagnosis.ConclusionsEShp is rare in individuals without MD, supporting its specificity and potential role in MD pathophysiology. The ATVA marker is reliable and reproducible and may serve as a CT biomarker for the hypoplastic MD endophenotype.</p>\",\"PeriodicalId\":49960,\"journal\":{\"name\":\"Journal of Vestibular Research-Equilibrium & Orientation\",\"volume\":\" \",\"pages\":\"9574271251371541\"},\"PeriodicalIF\":3.3000,\"publicationDate\":\"2025-08-25\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Vestibular Research-Equilibrium & Orientation\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1177/09574271251371541\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"NEUROSCIENCES\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Vestibular Research-Equilibrium & Orientation","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1177/09574271251371541","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"NEUROSCIENCES","Score":null,"Total":0}
Prevalence of the endolymphatic sac hypoplasia in a cohort of individuals without Meniere disease.
PurposeTo estimate the prevalence of endolymphatic sac hypoplasia (EShp)-a proposed specific finding in Ménière's disease (MD) that defines an endophenotype characterized by bilateral involvement, male predominance, temporal bone abnormalities, and familial clustering-in individuals without MD, to assess its specificity for the condition.MethodsWe analyzed 956 temporal bone CT scans from individuals without MD to assess the prevalence of EShp using the Angular Trajectory of the Vestibular Aqueduct (ATVA) marker. ATVA distribution, reproducibility, and associations with clinical variables were also evaluated.ResultsEShp was identified in 6 ears from 4 individuals, yielding a prevalence of 0.6% per patient and 0.8% per ear. ATVA values had a median of 95.0° (IQR = 12.5°, range 65.9°-159.4°). Interobserver agreement was good (ICC = 0.75), with a mean bias of 6.2° ± 5.4° and 5.5% of ears outside the 95% limits of agreement. No significant associations were found between ATVA and sex, age, or clinical diagnosis.ConclusionsEShp is rare in individuals without MD, supporting its specificity and potential role in MD pathophysiology. The ATVA marker is reliable and reproducible and may serve as a CT biomarker for the hypoplastic MD endophenotype.
期刊介绍:
Journal of Vestibular Research is a peer-reviewed journal that publishes experimental and observational studies, review papers, and theoretical papers based on current knowledge of the vestibular system. Subjects of the studies can include experimental animals, normal humans, and humans with vestibular or other related disorders. Study topics can include the following:
Anatomy of the vestibular system, including vestibulo-ocular, vestibulo-spinal, and vestibulo-autonomic pathways
Balance disorders
Neurochemistry and neuropharmacology of balance, both at the systems and single neuron level
Neurophysiology of balance, including the vestibular, ocular motor, autonomic, and postural control systems
Psychophysics of spatial orientation
Space and motion sickness
Vestibular rehabilitation
Vestibular-related human performance in various environments