{"@context":{"@vocab":"https://cir.nii.ac.jp/schema/1.0/","rdfs":"http://www.w3.org/2000/01/rdf-schema#","dc":"http://purl.org/dc/elements/1.1/","dcterms":"http://purl.org/dc/terms/","foaf":"http://xmlns.com/foaf/0.1/","prism":"http://prismstandard.org/namespaces/basic/2.0/","cinii":"http://ci.nii.ac.jp/ns/1.0/","datacite":"https://schema.datacite.org/meta/kernel-4/","ndl":"http://ndl.go.jp/dcndl/terms/","jpcoar":"https://github.com/JPCOAR/schema/blob/master/2.0/"},"@id":"https://cir.nii.ac.jp/crid/1390282679749905280.json","@type":"Article","productIdentifier":[{"identifier":{"@type":"DOI","@value":"10.11289/otoljpn.21.808"}},{"identifier":{"@type":"NAID","@value":"10030285311"}},{"identifier":{"@type":"URI","@value":"https://search.jamas.or.jp/link/ui/2012102246"}}],"dc:title":[{"@language":"en","@value":"MPO-ANCA-associated middle ear disease with sensorineural hearing loss and facial palsy: case report, review of literature, proposed diagnostic criteria"},{"@language":"ja","@value":"感音難聴と顔面神経麻痺を示すMPO-ANCA関連中耳疾患の1例: 診断基準案の提案"}],"dc:language":"ja","description":[{"type":"abstract","notation":[{"@language":"en","@value":"We reported a case of MPO-ANCA-associated middle ear disease with sensorineural hearing loss and facial palsy.  A 62-year-old female first complained of bilateral aural obstruction and the treatment for otitis media with effusion was ineffective.  Then, bilateral sensorineural hearing loss developed, and after the transient effect of steroid therapy, left facial palsy appeared.  CT demonstrated that the low density area in the middle ear cavity without bone destruction, and its granular tissue surgically resected showed non-specific findings by the histopathological examination.  MPO-ANCA was positive, but PR3-ANCA was negative in the serum without pulmonary and renal lesions.  Steroid treatment with cyclophosphamaide improved the hearing loss and facial palsy with a decrease of serum MPO-ANCA level.<br> Although it was reported previously that patients with MPO-ANCA-associated middle ear disease with sensorineural hearing loss and facial palsy were successfully treated by combination therapy with steroid and immunosuppressive drug in addition to our case, they could not meet the criteria of ANCA-associated vasculitides such as Wegener's glanulomatosis.  Therefore, we proposed the diagnostic term and criteria of MPO-ANCA-associated middle ear disease with sensorineural hearing loss and facial palsy.  The underlying pathophysiology of this disease may be MPO-ANCA-induced vasculitis, but review of literature showed that its clinical characteristics are different from those of ANCA-associated vasculitides.<br>The proposed diagnostic criteria were as follows: 1) the onset of otitis media-like symptom, 2) progressed sensorineural hearing loss, 3) complicating facial palsy, 4) lesion limited to the middle ear without pulmonary and renal lesions, 5) MPO-ANCA positive, but PR3-ANCA negative in the serum, 6) no findings of vasculitis in the middle ear lesion."},{"@language":"ja","@value":"感音難聴と顔面神経麻痺を示したMPO-ANCA関連中耳疾患症例を報告した。耳閉感で初発し、滲出性中耳炎の治療を受けるも両感音難聴が進行した。ステロイドの効果は一時的であり、その後、左顔面神経麻痺が出現した。側頭骨CTでは中耳に軟部陰影を認めたが、骨破壊なし。PR3-ANCA陰性、MPO-ANCA陽性であったが、肺・腎病変はなく、中耳組織に血管炎の所見を認めなかった。ステロイドと免疫抑制薬の投与により、難聴と顔面神経麻痺は改善した。このような感音難聴と顔面神経麻痺を示すMPO-ANCA関連中耳疾患は、MPO-ANCAによる血管炎が関与している可能性があるが、組織検査でANCA関連血管炎と診断できない。また、本疾患はANCA関連血管炎とは臨床的特徴が異なっていた。本疾患はステロイドと免疫抑制薬の併用治療が有効であるため、免疫抑制剤の使用の根拠となる診断名が必要と考え、診断基準とともに提案した。"}],"abstractLicenseFlag":"disallow"}],"creator":[{"@id":"https://cir.nii.ac.jp/crid/1420845751163973888","@type":"Researcher","personIdentifier":[{"@type":"KAKEN_RESEARCHERS","@value":"60380028"},{"@type":"NRID","@value":"1000060380028"},{"@type":"NRID","@value":"9000003770100"},{"@type":"NRID","@value":"9000410657714"},{"@type":"NRID","@value":"9000257948549"},{"@type":"NRID","@value":"9000020926445"},{"@type":"NRID","@value":"9000311063117"},{"@type":"NRID","@value":"9000283281486"},{"@type":"NRID","@value":"9000258225930"},{"@type":"NRID","@value":"9000311063242"},{"@type":"NRID","@value":"9000413900873"},{"@type":"NRID","@value":"9000283104863"},{"@type":"NRID","@value":"9000261039186"},{"@type":"NRID","@value":"9000404787283"},{"@type":"NRID","@value":"9000399542436"},{"@type":"NRID","@value":"9000292183068"},{"@type":"NRID","@value":"9000283315201"},{"@type":"NRID","@value":"9000021554244"},{"@type":"NRID","@value":"9000243896246"},{"@type":"NRID","@value":"9000398302593"},{"@type":"NRID","@value":"9000412185508"},{"@type":"NRID","@value":"9000329015030"},{"@type":"NRID","@value":"9000412234239"},{"@type":"NRID","@value":"9000399773423"},{"@type":"NRID","@value":"9000410657715"},{"@type":"NRID","@value":"9000415417406"},{"@type":"NRID","@value":"9000370764773"},{"@type":"NRID","@value":"9000408919303"},{"@type":"NRID","@value":"9000257947990"},{"@type":"NRID","@value":"9000399773415"},{"@type":"NRID","@value":"9000292185287"},{"@type":"RESEARCHMAP","@value":"https://researchmap.jp/ykitamura1212"}],"foaf:name":[{"@language":"en","@value":"Kitamura Yoshiaki"},{"@language":"ja","@value":"北村 嘉章"}],"jpcoar:affiliationName":[{"@language":"en","@value":"Department of Otolaryngology, University of Tokushima School of Medicine"},{"@language":"ja","@value":"徳島大学医学部耳鼻咽喉科学教室"}]},{"@id":"https://cir.nii.ac.jp/crid/1410009222134030213","@type":"Researcher","personIdentifier":[{"@type":"NRID","@value":"9000003746303"}],"foaf:name":[{"@language":"en","@value":"Abe Koji"},{"@language":"ja","@value":"阿部 晃治"}],"jpcoar:affiliationName":[{"@language":"ja","@value":"徳島大学医学部耳鼻咽喉科学教室"},{"@language":"en","@value":"Department of Otolaryngology, University of Tokushima School of Medicine"}]},{"@id":"https://cir.nii.ac.jp/crid/1410009222134030208","@type":"Researcher","personIdentifier":[{"@type":"NRID","@value":"9000000832544"}],"foaf:name":[{"@language":"en","@value":"Endo Aki"},{"@language":"ja","@value":"遠藤 亜紀"}],"jpcoar:affiliationName":[{"@language":"en","@value":"Department of Otolaryngology, Shikoku Central Hospital"},{"@language":"ja","@value":"四国中央病院耳鼻咽喉科"}]},{"@id":"https://cir.nii.ac.jp/crid/1410009222134030210","@type":"Researcher","personIdentifier":[{"@type":"NRID","@value":"9000019065000"}],"foaf:name":[{"@language":"en","@value":"Kamimura Seiichiro"},{"@language":"ja","@value":"神村 盛一郎"}],"jpcoar:affiliationName":[{"@language":"en","@value":"Department of Otolaryngology, University of Tokushima School of Medicine"},{"@language":"ja","@value":"徳島大学医学部耳鼻咽喉科学教室"}]},{"@id":"https://cir.nii.ac.jp/crid/1410009222134030211","@type":"Researcher","personIdentifier":[{"@type":"NRID","@value":"9000000433511"}],"foaf:name":[{"@language":"en","@value":"Tamura Koichi"},{"@language":"ja","@value":"田村 公一"}],"jpcoar:affiliationName":[{"@language":"ja","@value":"徳島大学医学部耳鼻咽喉科学教室"},{"@language":"en","@value":"Department of Otolaryngology, University of Tokushima School of 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Noriaki"},{"@language":"ja","@value":"武田 憲昭"}],"jpcoar:affiliationName":[{"@language":"en","@value":"Department of Otolaryngology, University of Tokushima School of Medicine"},{"@language":"ja","@value":"徳島大学医学部耳鼻咽喉科学教室"}]},{"@id":"https://cir.nii.ac.jp/crid/1410009222134030212","@type":"Researcher","personIdentifier":[{"@type":"NRID","@value":"9000000832567"}],"foaf:name":[{"@language":"en","@value":"Udaka Jiro"},{"@language":"ja","@value":"宇高 二良"}],"jpcoar:affiliationName":[{"@language":"en","@value":"Udaka ENT Clinic"},{"@language":"ja","@value":"宇高耳鼻咽喉科医院"}]},{"@id":"https://cir.nii.ac.jp/crid/1410001204845768960","@type":"Researcher","personIdentifier":[{"@type":"NRID","@value":"9000004456133"}],"foaf:name":[{"@language":"en","@value":"Jinnouchi Osamu"},{"@language":"ja","@value":"陣内 自治"}],"jpcoar:affiliationName":[{"@language":"en","@value":"Department of Otolaryngology, University of Tokushima School of Medicine"},{"@language":"ja","@value":"徳島大学医学部耳鼻咽喉科学教室"}]}],"publication":{"publicationIdentifier":[{"@type":"PISSN","@value":"09172025"},{"@type":"EISSN","@value":"18841457"},{"@type":"NCID","@value":"AN10358085"}],"prism:publicationName":[{"@language":"en","@value":"Otology Japan"},{"@language":"ja","@value":"Ｏｔｏｌｏｇｙ　Ｊａｐａｎ"},{"@language":"en","@value":"Otol Jpn"},{"@language":"ja","@value":"Ｏｔｏｌ　Ｊｐｎ"}],"dc:publisher":[{"@language":"en","@value":"Japan Otological Society"},{"@language":"ja","@value":"一般社団法人　日本耳科学会"}],"prism:publicationDate":"2011","prism:volume":"21","prism:number":"5","prism:startingPage":"808","prism:endingPage":"815"},"url":[{"@id":"https://search.jamas.or.jp/link/ui/2012102246"}],"availableAt":"2011","foaf:topic":[{"@id":"https://cir.nii.ac.jp/all?q=MPO-ANCA-associated%20vasculitides","dc:title":"MPO-ANCA-associated vasculitides"},{"@id":"https://cir.nii.ac.jp/all?q=Wegener's%20glanulomatosis","dc:title":"Wegener's glanulomatosis"},{"@id":"https://cir.nii.ac.jp/all?q=microscopic%20polyangitis","dc:title":"microscopic polyangitis"},{"@id":"https://cir.nii.ac.jp/all?q=Churg-Strauss%20syndrome","dc:title":"Churg-Strauss syndrome"},{"@id":"https://cir.nii.ac.jp/all?q=MPO-ANCA-associated%20hypertrophic%20cranial%20pachymenigitis","dc:title":"MPO-ANCA-associated hypertrophic cranial pachymenigitis"},{"@id":"https://cir.nii.ac.jp/all?q=MPO-ANCA%E9%96%A2%E9%80%A3%E8%A1%80%E7%AE%A1%E7%82%8E","dc:title":"MPO-ANCA関連血管炎"},{"@id":"https://cir.nii.ac.jp/all?q=%E3%82%A6%E3%82%A8%E3%82%B2%E3%83%8A%E3%83%BC%E8%82%89%E8%8A%BD%E8%85%AB%E7%97%87","dc:title":"ウエゲナー肉芽腫症"},{"@id":"https://cir.nii.ac.jp/all?q=%E9%A1%95%E5%BE%AE%E9%8F%A1%E7%9A%84%E5%A4%9A%E7%99%BA%E8%A1%80%E7%AE%A1%E7%82%8E","dc:title":"顕微鏡的多発血管炎"},{"@id":"https://cir.nii.ac.jp/all?q=%E3%82%A2%E3%83%AC%E3%83%AB%E3%82%AE%E3%83%BC%E6%80%A7%E8%82%89%E8%8A%BD%E8%85%AB%E6%80%A7%E8%A1%80%E7%AE%A1%E7%82%8E","dc:title":"アレルギー性肉芽腫性血管炎"},{"@id":"https://cir.nii.ac.jp/all?q=MPO-ANCA%E9%99%BD%E6%80%A7%E8%82%A5%E5%8E%9A%E6%80%A7%E7%A1%AC%E8%86%9C%E7%82%8E","dc:title":"MPO-ANCA陽性肥厚性硬膜炎"}],"relatedProduct":[{"@id":"https://cir.nii.ac.jp/crid/1360011145808130176","@type":"Article","relationType":["cites"],"jpcoar:relatedTitle":[{"@value":"Nomenclature of Systemic 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レイ"}]},{"@id":"https://cir.nii.ac.jp/crid/1390001204771932032","@type":"Article","relationType":["isCitedBy"],"jpcoar:relatedTitle":[{"@language":"ja","@value":"ANCA関連血管炎による難治性中耳炎の検討"},{"@language":"en","@value":"Clinical analysis of refractory otitis media due to ANCA related vasculitis"}]},{"@id":"https://cir.nii.ac.jp/crid/1390001204771936768","@type":"Article","relationType":["isCitedBy"],"jpcoar:relatedTitle":[{"@language":"ja","@value":"耳症状を初発としたANCA関連血管炎性中耳炎症例の臨床像"},{"@language":"en","@value":"Clinical features of ANCA-associated vasculitis initially diagnosed by otologic symptoms"}]},{"@id":"https://cir.nii.ac.jp/crid/1390282679236505856","@type":"Article","relationType":["cites"],"jpcoar:relatedTitle":[{"@language":"en","@value":"A Study of C-ANCA Negative Wegener's Granulomatosis."},{"@value":"Ｃ‐ＡＮＣＡ陰性Ｗｅｇｅｎｅｒ肉芽腫症の検討"}]},{"@id":"https://cir.nii.ac.jp/crid/1390282679239203712","@type":"Article","relationType":["cites"],"jpcoar:relatedTitle":[{"@language":"en","@value":"A Case of Fluctuating Sensorineural Hearing Loss Associated with Microscopic Polyangitis"},{"@value":"顕微鏡的多発血管炎に伴った変動する感音難聴例"},{"@language":"ja-Kana","@value":"リンショウ ケンビキョウテキ タハツ ケッカンエン ニ トモナッタ ヘンドウ スル カンオン ナンチョウレイ"}]},{"@id":"https://cir.nii.ac.jp/crid/1390282679240139904","@type":"Article","resourceType":"学術雑誌論文(journal article)","relationType":["cites"],"jpcoar:relatedTitle":[{"@language":"en","@value":"Wegener's Granulomatosis with Otolaryngological Symptoms"},{"@value":"ウェゲナー肉芽腫症１６例の臨床的検討"},{"@language":"ja-Kana","@value":"リンショウ ウェゲナー ニクゲシュショウ 16レイ ノ リンショウテキ ケントウ"}]},{"@id":"https://cir.nii.ac.jp/crid/1390282679241347072","@type":"Article","relationType":["cites"],"jpcoar:relatedTitle":[{"@language":"en","@value":"A Case of P-ANCA Positive Wegener's Granulomatosis with Progressive Hearing Loss"},{"@language":"ja","@value":"進行性難聴を呈したＰ‐ＡＮＣＡ陽性Ｗｅｇｅｎｅｒ肉芽腫症"},{"@value":"臨床 進行性難聴を呈したP-ANCA陽性Wegener肉芽腫症"},{"@language":"ja-Kana","@value":"リンショウ シンコウセイ ナンチョウ オ テイシタ P ANCA ヨウセイ Wegener 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