神戸大学附属図書館デジタルアーカイブ
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https://hdl.handle.net/20.500.14094/90007067
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2024-04-27
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90007067 (fulltext)
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メタデータID
90007067
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open access
出版タイプ
Version of Record
タイトル
Vasculitis with superior ophthalmic vein thrombosis compatible with neuro-neutrophilic disease
著者
Sakamoto, Mari ; Kurimoto, Takuji ; Mori, Sotaro ; Ueda, Kaori ; Keshi, Yukako ; Yamada, Yuko ; Azumi, Atsushi ; Shimono, Taro ; Nakamura, Makoto
著者ID
A2431
KUID
https://kuid-rm-web.ofc.kobe-u.ac.jp/search/detail?systemId=e0fd31caf46bb45b520e17560c007669
著者名
Sakamoto, Mari
坂本, 麻里
サカモト, マリ
所属機関名
医学研究科
著者ID
A1535
研究者ID
1000050388815
KUID
https://kuid-rm-web.ofc.kobe-u.ac.jp/search/detail?systemId=09106101bc83c492520e17560c007669
著者名
Kurimoto, Takuji
栗本, 拓治
クリモト, タクジ
所属機関名
医学研究科
著者名
Mori, Sotaro
著者ID
A2426
研究者ID
1000070792232
KUID
https://kuid-rm-web.ofc.kobe-u.ac.jp/search/detail?systemId=121a64d967b801de520e17560c007669
著者名
Ueda, Kaori
上田, 香織
ウエダ, カオリ
所属機関名
医学部附属病院
著者名
Keshi, Yukako
著者ID
A1365
研究者ID
1000000359861
KUID
https://kuid-rm-web.ofc.kobe-u.ac.jp/search/detail?systemId=d9c758f40b2218da520e17560c007669
著者名
Yamada, Yuko
山田, 裕子
ヤマダ, ユウコ
所属機関名
医学研究科
著者名
Azumi, Atsushi
著者名
Shimono, Taro
著者ID
A0882
研究者ID
1000080273788
KUID
https://kuid-rm-web.ofc.kobe-u.ac.jp/search/detail?systemId=82142348522a81ad520e17560c007669
著者名
Nakamura, Makoto
中村, 誠
ナカムラ, マコト
所属機関名
医学研究科
収録物名
American Journal of Ophthalmology Case Reports
巻(号)
12
ページ
39-44
出版者
Elsevier
刊行日
2018-12
公開日
2020-04-30
抄録
Purpose: To present a unique case of neuro-neutrophilic disease with inflammation and thrombosis of the superior ophthalmic vein (SOV). Observations: A 43-year-old Japanese man with past histories of oculomotor paralysis, auditory disorder, ischemic enteritis, and recurrent oral ulceration was referred to our hospital because of blurred vision in his right eye. Ophthalmic examination revealed decreased best corrected visual acuity and central scotoma in his right eye. Orbit magnetic resonance imaging (MRI) revealed an enlarged SOV in the right eye, with Gadolinium (Gd) enhancement in the wall of the vein but not inside the vein, indicating thrombosis. Multiple Gd-enhanced hyperintense lesions were also observed in the juxtacortical area of the brain. We diagnosed the patient with vasculitis in the right SOV that was adversely affecting the optic nerve. We ruled out systemic thrombophilia, infections, and malignancy by systemic examinations. The human leukocyte antigen (HLA) typing was Cw1-, B54-, B61-, A2-, A24-, and DR4-positive and B51-negative. We treated the patient with systemic steroid and anticoagulant therapy. After three courses of steroid pulse therapy, his symptoms and the MRI findings of the right SOV and brain improved; therefore, we decided to discontinue the anticoagulant therapy. One month after anticoagulant cessation, MRI revealed recurrence of the thrombus and enlargement of the right SOV despite the lack of vision worsening. We restarted the anticoagulant therapy while continuing the oral prednisolone treatment. At the final visit, 14 months after the onset of the disease, the patient was still receiving oral anticoagulation with warfarin potassium and prednisolone (5 mg/day). His symptoms and the right eye's visual function remained normal with a mildly enlarged SOV; there was less Gd enhancement and no brain lesions on MRI. Conclusions and importance: We treated a unique case of possible neuro-neutrophilic disease that presented visual disturbances due to right SOV inflammation and thrombosis. Anticoagulation and systemic steroid therapies were required to reduce the inflammation and to prevent the recurrence of thrombosis.
キーワード
Neuro-neutrophilic disease
Neuro-Sweet disease
Neuro-Behçet disease
Thrombosis
Human leukocyte antigen
カテゴリ
医学研究科
医学部附属病院
学術雑誌論文
権利
© 2018 The Authors. Published by Elsevier Inc.
This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
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資源タイプ
journal article
言語
English (英語)
eISSN
2451-9936
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関連情報
DOI
https://doi.org/10.1016/j.ajoc.2018.08.005
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