{"_buckets": {"deposit": "f6a4cc96-61ab-4ef4-a53b-6943c0ea899c"}, "_deposit": {"id": "21405", "owners": [], "pid": {"revision_id": 0, "type": "depid", "value": "21405"}, "status": "published"}, "_oai": {"id": "oai:soar-ir.repo.nii.ac.jp:00021405", "sets": []}, "author_link": ["109532"], "item_14_biblio_info_6": {"attribute_name": "書誌情報", "attribute_value_mlt": [{"bibliographicIssueDates": {"bibliographicIssueDate": "2019-03-31", "bibliographicIssueDateType": "Issued"}}]}, "item_14_date_granted_8": {"attribute_name": "学位授与年月日", "attribute_value_mlt": [{"subitem_dategranted": "2019-03-31"}]}, "item_14_degree_grantor_10": {"attribute_name": "学位授与機関", "attribute_value_mlt": [{"subitem_degreegrantor": [{"subitem_degreegrantor_name": "信州大学(Shinshu university)"}], "subitem_degreegrantor_identifier": [{"subitem_degreegrantor_identifier_name": "13601", "subitem_degreegrantor_identifier_scheme": "kakenhi"}]}]}, "item_14_degree_name_9": {"attribute_name": "学位名", "attribute_value_mlt": [{"subitem_degreename": "博士（医学）"}]}, "item_14_description_26": {"attribute_name": "内容記述", "attribute_value_mlt": [{"subitem_description": "雑誌に発表。EUROPEAN JOURNAL OF NEUROLOGY. 27(2):369-375 (2020); doi:10.1111/ene.14086.", "subitem_description_type": "Other"}]}, "item_14_description_37": {"attribute_name": "資源タイプ（コンテンツの種類）", "attribute_value_mlt": [{"subitem_description": "Thesis", "subitem_description_type": "Other"}]}, "item_14_description_5": {"attribute_name": "引用", "attribute_value_mlt": [{"subitem_description": "近藤　恭史. Factors predictive of the presence of a CSF1R mutation in patients with leukoencephalopathy （白質異常症におけるCSF1R遺伝子変異陽性を予測する患者要因の検討）. 信州大学, 2019, 博士論文.", "subitem_description_type": "Other"}]}, "item_14_dissertation_number_7": {"attribute_name": "学位授与番号", "attribute_value_mlt": [{"subitem_dissertationnumber": "13601甲第1195号"}]}, "item_14_link_74": {"attribute_name": "WoS", "attribute_value_mlt": [{"subitem_link_text": "Web of Science", "subitem_link_url": "http://gateway.isiknowledge.com/gateway/Gateway.cgi?\u0026GWVersion=2\u0026SrcAuth=ShinshuUniv\u0026SrcApp=ShinshuUniv\u0026DestLinkType=FullRecord\u0026DestApp=WOS\u0026KeyUT=000505919500023"}]}, "item_14_publisher_4": {"attribute_name": "出版者", "attribute_value_mlt": [{"subitem_publisher": "信州大学"}]}, "item_14_relation_54": {"attribute_name": "PubMed", "attribute_value_mlt": [{"subitem_relation_name": [{"subitem_relation_name_text": "31520500"}], "subitem_relation_type_id": {"subitem_relation_type_id_text": "https://pubmed.ncbi.nlm.nih.gov/31520500/", "subitem_relation_type_select": "PMID"}}]}, "item_14_relation_55": {"attribute_name": "DOI", "attribute_value_mlt": [{"subitem_relation_name": [{"subitem_relation_name_text": "10.1111/ene.14086"}], "subitem_relation_type_id": {"subitem_relation_type_id_text": "https://doi.org/10.1111/ene.14086", "subitem_relation_type_select": "DOI"}}]}, "item_14_rights_69": {"attribute_name": "権利", "attribute_value_mlt": [{"subitem_rights": "© 2019 The Authors. European Journal of Neurology published by John Wiley \u0026 Sons Ltd on behalf of European Academy of Neurology \nThis is an open access article under the terms of the Creative Commons Attribution‐NonCommercial‐NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.\n"}]}, "item_14_select_71": {"attribute_name": "著者版フラグ", "attribute_value_mlt": [{"subitem_select_item": "ETD"}]}, "item_14_text_11": {"attribute_name": "学位の区分", "attribute_value_mlt": [{"subitem_text_value": "doctoral"}]}, "item_14_text_12": {"attribute_name": "学位の分野", "attribute_value_mlt": [{"subitem_text_value": "医学"}]}, "item_14_text_13": {"attribute_name": "学位の報告番号", "attribute_value_mlt": [{"subitem_text_value": "甲第1195号"}]}, "item_14_text_76": {"attribute_name": "wosonly authkey", "attribute_value_mlt": [{"subitem_text_value": "adult-onset leukoencephalopathy with axonal spheroids and pigmented glia; colony-stimulating factor 1 receptor; leukoencephalopathy; logistic regression; diagnostic criteria"}]}, "item_14_text_77": {"attribute_name": "wosonly keywords", "attribute_value_mlt": [{"subitem_text_value": "HEREDITARY DIFFUSE LEUKOENCEPHALOPATHY; ADULT-ONSET LEUKOENCEPHALOPATHY; AXONAL SPHEROIDS; PIGMENTED GLIA; HDLS; POLD"}]}, "item_14_textarea_75": {"attribute_name": "wosonly abstract", "attribute_value_mlt": [{"subitem_textarea_value": "Background and purpose The purpose was to identify statistically factors that correlate with the presence of a colony-stimulating factor 1 receptor (CSF1R) mutation and to reevaluate the accuracy of the current diagnostic criteria for CSF1R-related leukoencephalopathy. Methods CSF1R testing was conducted on 145 consecutive leukoencephalopathy cases who were clinically suspected of having adult-onset leukoencephalopathy with axonal spheroids and pigmented glia. From these, 135 cases whose detailed clinical information was available were enrolled. Forward logistic stepwise regression was performed to generate a probability model to predict a positive CSF1R mutation result. The current diagnostic criteria were also applied to our cohort and their sensitivity and specificity were calculated. Results Twenty-eight CSF1R-mutation-positive cases and 107 CSF1R-mutation-negative cases were identified. Our probability model suggested that factors raising the probability of a CSF1R-mutation-positive result were younger onset, parkinsonism, thinning of the corpus callosum and diffusion-restricted lesions. It also showed that involuntary movements and brainstem or cerebellar atrophy were negative predictors of a CSF1R-mutation-positive result. In our cohort, the sensitivity and specificity for \u0027probable\u0027 or \u0027possible\u0027 CSF1R-related leukoencephalopathy were 81% and 14%, respectively. Conclusions Clinical and brain imaging features predictive of the presence of a CSF1R mutation are proposed. Consideration of these factors will help prioritize patients for CSF1R testing."}]}, "item_1627890986942": {"attribute_name": "出版タイプ", "attribute_value_mlt": [{"subitem_version_resource": "http://purl.org/coar/version/c_be7fb7dd8ff6fe43", "subitem_version_type": "NA"}]}, "item_access_right": {"attribute_name": "アクセス権", "attribute_value_mlt": [{"subitem_access_right": "open access", "subitem_access_right_uri": "http://purl.org/coar/access_right/c_abf2"}]}, "item_creator": {"attribute_name": "著者", "attribute_type": "creator", "attribute_value_mlt": [{"creatorNames": [{"creatorName": "近藤, 恭史", "creatorNameLang": "ja"}], "nameIdentifiers": [{"nameIdentifier": "109532", "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": "2020-06-12"}], "displaytype": "detail", "download_preview_message": "", "file_order": 0, "filename": "15MH202J_yoshi.pdf", "filesize": [{"value": "147.1 kB"}], "format": "application/pdf", "future_date_message": "", "is_thumbnail": false, "licensetype": "license_note", "mimetype": "application/pdf", "size": 147100.0, "url": {"label": "内容の要旨", "url": "https://soar-ir.repo.nii.ac.jp/record/21405/files/15MH202J_yoshi.pdf"}, "version_id": "0099ccf4-a95c-4329-bd6f-e13438bb73bb"}, {"accessrole": "open_date", "date": [{"dateType": "Available", "dateValue": "2020-06-12"}], "displaytype": "detail", "download_preview_message": "", "file_order": 1, "filename": "15MH202J_shinsa.pdf", "filesize": [{"value": "101.5 kB"}], "format": "application/pdf", "future_date_message": "", "is_thumbnail": false, "licensetype": "license_note", "mimetype": "application/pdf", "size": 101500.0, "url": {"label": "審査結果の要旨", "url": "https://soar-ir.repo.nii.ac.jp/record/21405/files/15MH202J_shinsa.pdf"}, "version_id": "23c20c8b-69fb-498d-a92d-eeec5fb8fe6d"}, {"accessrole": "open_date", "date": [{"dateType": "Available", "dateValue": "2020-06-12"}], "displaytype": "detail", "download_preview_message": "", "file_order": 2, "filename": "15MH202J_ronbun.pdf", "filesize": [{"value": "532.9 kB"}], "format": "application/pdf", "future_date_message": "", "is_thumbnail": false, "licensefree": "© 2019 The Authors. European Journal of Neurology published by John Wiley \u0026 Sons Ltd on behalf of European Academy of Neurology \nThis is an open access article under the terms of the Creative Commons Attribution‐NonCommercial‐NoDerivs License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.\n", "licensetype": "license_note", "mimetype": "application/pdf", "size": 532900.0, "url": {"label": "博士論文の全文", "objectType": "fulltext", "url": "https://soar-ir.repo.nii.ac.jp/record/21405/files/15MH202J_ronbun.pdf"}, "version_id": "ff99c723-750e-432f-9fef-5573dc6c761f"}]}, "item_language": {"attribute_name": "言語", "attribute_value_mlt": [{"subitem_language": "eng"}]}, "item_resource_type": {"attribute_name": "資源タイプ", "attribute_value_mlt": [{"resourcetype": "doctoral thesis", "resourceuri": "http://purl.org/coar/resource_type/c_db06"}]}, "item_title": "Factors predictive of the presence of a CSF1R mutation in patients with leukoencephalopathy （白質異常症におけるCSF1R遺伝子変異陽性を予測する患者要因の検討）", "item_titles": {"attribute_name": "タイトル", "attribute_value_mlt": [{"subitem_title": "Factors predictive of the presence of a CSF1R mutation in patients with leukoencephalopathy （白質異常症におけるCSF1R遺伝子変異陽性を予測する患者要因の検討）", "subitem_title_language": "en"}]}, "item_type_id": "14", "owner": "1", "path": ["849"], "permalink_uri": "http://hdl.handle.net/10091/00022162", "pubdate": {"attribute_name": "PubDate", "attribute_value": "2020-06-15"}, "publish_date": "2020-06-15", "publish_status": "0", "recid": "21405", "relation": {}, "relation_version_is_last": true, "title": ["Factors predictive of the presence of a CSF1R mutation in patients with leukoencephalopathy （白質異常症におけるCSF1R遺伝子変異陽性を予測する患者要因の検討）"], "weko_shared_id": -1}