{"created":"2021-03-01T06:31:25.770558+00:00","id":25905,"links":{},"metadata":{"_buckets":{"deposit":"f7b049b1-1ce8-48f1-89d4-ab9437ea49cc"},"_deposit":{"id":"25905","owners":[],"pid":{"revision_id":0,"type":"depid","value":"25905"},"status":"published"},"_oai":{"id":"oai:niigata-u.repo.nii.ac.jp:00025905","sets":["432:1291:1292:1324","453:456"]},"item_7_alternative_title_1":{"attribute_name":"その他のタイトル","attribute_value_mlt":[{"subitem_alternative_title":"Basal Cell nevoid Syndrome - Report of a case with 20 years' folow-up -"}]},"item_7_biblio_info_6":{"attribute_name":"書誌情報","attribute_value_mlt":[{"bibliographicIssueDates":{"bibliographicIssueDate":"1997-07","bibliographicIssueDateType":"Issued"},"bibliographicIssueNumber":"1","bibliographicPageEnd":"28","bibliographicPageStart":"23","bibliographicVolumeNumber":"27","bibliographic_titles":[{"bibliographic_title":"新潟歯学会雑誌"},{"bibliographic_title":"新潟歯学会雑誌","bibliographic_titleLang":"en"}]}]},"item_7_description_4":{"attribute_name":"抄録","attribute_value_mlt":[{"subitem_description":"Basal cell nevoid syndrome is an autosomal dominant herediatry disease with multiple pits on palmar and plantar skin, bifid rib, ocular hyperterolism, multiple jaw cysts and many other malformations. Most of the jaw cysts found in this syndrome are odontogenic keratocysts. In this paper, 11-year-old boy with basal cell nevoid syndrome who was followed up for 20 years was reported. Clinical examination showed that he had ocular hypertelorism, broad nasal root, temporal and frontal bossings, mandibular prognathism, and diffuse swelling in the right cheek. His chest radiograph showed clavate lib and remaining of 6th cervical lib. Panoramic radiograph showed many cystic lesions in the maxilla and mandible. Analogous findings were also observed in his father and brother. The cysts were treated by marsupialization, enuculation and primary closure, and/or enucleation and packing open. Recurrences occurred three times in two cysts with bony defects during the whereas three new cysts developed period of 20 years. One of the new cysts was discovered 19 years after his first visit. Regrowth after marsupialization was encountered in two cysts with epithelial islands. Histologically, all cysts were odontogenic keratocysts and there were epithelial islands in the cyst walls in three cysts. A long-term observation is needed because of the high rate of recurrence and development of new cysts in patients with basal cell nevoid syndrome.","subitem_description_type":"Abstract"},{"subitem_description":"基底細胞母斑症候群は皮膚病変として掌蹠の小窩、二分肋骨、両眼隔離、前頭及び側頭の突出、多発性顎嚢胞や他の様々な異常を伴う常染色体優勢遺伝の疾患である｡本症候群で発生する顎嚢胞はほとんどが歯原性角化嚢胞(以下OKC)である｡今回我々は20年に渡って観察した基底細胞母斑症候群の11才の男児の1例を報告した｡初診時現症として両眼隔離、鼻根の低下、前頭及び側頭部の突出、下顎前突を認めた｡胸部Ⅹ線で肋骨の梶棒状変化、第6頚肋骨の残存を認め、パノラマX線写真では上下顎に多発性の嚢胞様骨透過像を認めた｡同様の所見は患者の父親と兄にも認められた。全ての嚢胞に対し開窓術、摘出開創術あるいは摘出-時閉鎖術を施行した。20年の経過観察中に二つの嚢胞に3回の再発と3回の新たな嚢胞の形成を認め、そのうち1つは初診から19年目に発見されたものもあった｡病理組織診断はいずれも歯原性角化嚢胞であった｡開創後の増大は上皮島の認められた二つの嚢胞で認められた｡病理組織学的にはすべての嚢胞は歯原性角化嚢胞で3個の嚢胞の嚢胞壁に上皮島が認められた｡基底細胞母斑症候群の患者においては、顎嚢胞の再発率が高いこと、新たな嚢胞の形成が認められることなどから長期に渡る経過観察が必要と思われる｡","subitem_description_type":"Abstract"}]},"item_7_full_name_3":{"attribute_name":"著者別名","attribute_value_mlt":[{"nameIdentifiers":[{"nameIdentifier":"152904","nameIdentifierScheme":"WEKO"}],"names":[{"name":"Niimi, Kanae"}]},{"nameIdentifiers":[{"nameIdentifier":"152905","nameIdentifierScheme":"WEKO"}],"names":[{"name":"Hoshina, Yukiko"}]},{"nameIdentifiers":[{"nameIdentifier":"152906","nameIdentifierScheme":"WEKO"}],"names":[{"name":"Suzuki, Ichiro"}]},{"nameIdentifiers":[{"nameIdentifier":"152907","nameIdentifierScheme":"WEKO"}],"names":[{"name":"Nakajima, Tamio"}]}]},"item_7_publisher_7":{"attribute_name":"出版者","attribute_value_mlt":[{"subitem_publisher":"新潟歯学会"}]},"item_7_select_19":{"attribute_name":"著者版フラグ","attribute_value_mlt":[{"subitem_select_item":"publisher"}]},"item_7_source_id_11":{"attribute_name":"書誌レコードID","attribute_value_mlt":[{"subitem_source_identifier":"AN0018300X","subitem_source_identifier_type":"NCID"}]},"item_7_source_id_9":{"attribute_name":"ISSN","attribute_value_mlt":[{"subitem_source_identifier":"03850153","subitem_source_identifier_type":"ISSN"}]},"item_creator":{"attribute_name":"著者","attribute_type":"creator","attribute_value_mlt":[{"creatorNames":[{"creatorName":"新美, 秦恵"}],"nameIdentifiers":[{"nameIdentifier":"152900","nameIdentifierScheme":"WEKO"}]},{"creatorNames":[{"creatorName":"星名, 由紀子"}],"nameIdentifiers":[{"nameIdentifier":"152901","nameIdentifierScheme":"WEKO"}]},{"creatorNames":[{"creatorName":"鈴木, 一郎"}],"nameIdentifiers":[{"nameIdentifier":"152902","nameIdentifierScheme":"WEKO"}]},{"creatorNames":[{"creatorName":"中島, 民雄"}],"nameIdentifiers":[{"nameIdentifier":"152903","nameIdentifierScheme":"WEKO"}]}]},"item_files":{"attribute_name":"ファイル情報","attribute_type":"file","attribute_value_mlt":[{"accessrole":"open_date","date":[{"dateType":"Available","dateValue":"2019-08-19"}],"displaytype":"detail","filename":"NS_27(1)_23-28.pdf","filesize":[{"value":"4.9 MB"}],"format":"application/pdf","licensetype":"license_note","mimetype":"application/pdf","url":{"label":"NS_27(1)_23-28.pdf","url":"https://niigata-u.repo.nii.ac.jp/record/25905/files/NS_27(1)_23-28.pdf"},"version_id":"af4bb194-6975-4218-a5be-bbfbe7e775da"}]},"item_keyword":{"attribute_name":"キーワード","attribute_value_mlt":[{"subitem_subject":"Basal cell nevoid syndrome","subitem_subject_scheme":"Other"},{"subitem_subject":"基底細胞母斑症候群","subitem_subject_scheme":"Other"},{"subitem_subject":"Recurrence","subitem_subject_scheme":"Other"},{"subitem_subject":"再発","subitem_subject_scheme":"Other"},{"subitem_subject":"Multiple jaw cysts","subitem_subject_scheme":"Other"},{"subitem_subject":"多発性顎囊胞","subitem_subject_scheme":"Other"},{"subitem_subject":"Long term follow-up","subitem_subject_scheme":"Other"},{"subitem_subject":"長期観察","subitem_subject_scheme":"Other"}]},"item_language":{"attribute_name":"言語","attribute_value_mlt":[{"subitem_language":"jpn"}]},"item_resource_type":{"attribute_name":"資源タイプ","attribute_value_mlt":[{"resourcetype":"departmental bulletin paper","resourceuri":"http://purl.org/coar/resource_type/c_6501"}]},"item_title":"基底細胞母斑症候群の1例 : 20年間の経過について","item_titles":{"attribute_name":"タイトル","attribute_value_mlt":[{"subitem_title":"基底細胞母斑症候群の1例 : 20年間の経過について"},{"subitem_title":"基底細胞母斑症候群の1例 : 20年間の経過について","subitem_title_language":"en"}]},"item_type_id":"7","owner":"1","path":["456","1324"],"pubdate":{"attribute_name":"公開日","attribute_value":"2013-08-22"},"publish_date":"2013-08-22","publish_status":"0","recid":"25905","relation_version_is_last":true,"title":["基底細胞母斑症候群の1例 : 20年間の経過について"],"weko_creator_id":"1","weko_shared_id":null},"updated":"2022-12-15T03:55:53.284040+00:00"}