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伴抗磷脂综合征的大疱性坏疽性脓皮病:一例报告及文献复习

Bullous Pyoderma Gangrenosum Associated with Antiphospholipid Syndrome: A Case Report and Literature Review.

作者信息

Al-Bahrani Mayar, Alwashahi Yousuf, Almoqbali Ahmed, Alajmi Asma

机构信息

Family Medicine General Foundation Program, Oman Medical Specialty Board, Muscat, Oman.

Department of Dermatology, Directorate of General Health North Al Batinah, Sohar, Oman.

出版信息

Oman Med J. 2024 Jul 31;39(4):e658. doi: 10.5001/omj.2024.26. eCollection 2024 Jul.

Abstract

We report a rare case of a middle-aged Omani woman who was known to have primary antiphospholipid syndrome, glucose-6-phosphate dehydrogenase deficiency, and iron deficiency anaemia. Cannulation attempts caused bulla which progressed to ulceration. A pathergy phenomenon with high suspicion of pyoderma gangrenosum was postulated. Management with corticosteroids, cyclosporin, and finally ustekinumab, a novel biologic agent, yielded a beneficial response.

摘要

我们报告了一例罕见病例,患者为一名中年阿曼女性,患有原发性抗磷脂综合征、葡萄糖-6-磷酸脱氢酶缺乏症和缺铁性贫血。插管尝试导致形成水疱,进而发展为溃疡。推测存在高度怀疑为坏疽性脓皮病的同形反应现象。使用皮质类固醇、环孢素,最终使用新型生物制剂乌司奴单抗进行治疗,取得了有益的效果。

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Ustekinumab for the treatment of recalcitrant pyoderma gangrenosum: A case report.乌司奴单抗治疗顽固性坏疽性脓皮病:一例报告
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本文引用的文献

1
Ustekinumab as adjuvant treatment for all pyoderma gangrenosum subtypes.乌司奴单抗作为所有坏疽性脓皮病亚型的辅助治疗。
J Dermatolog Treat. 2022 Jun;33(4):2386-2390. doi: 10.1080/09546634.2021.1937475. Epub 2021 Jun 14.
3
Recent advances in managing and understanding pyoderma gangrenosum.坏疽性脓皮病管理与认识的最新进展
F1000Res. 2019 Dec 12;8. doi: 10.12688/f1000research.19909.1. eCollection 2019.
4
Antiphospholipid syndrome.抗磷脂综合征。
Best Pract Res Clin Rheumatol. 2020 Feb;34(1):101463. doi: 10.1016/j.berh.2019.101463. Epub 2019 Dec 19.
6
Etiology and management of pyoderma gangrenosum: a comprehensive review.坏疽性脓皮病的病因和治疗:全面综述。
Am J Clin Dermatol. 2012 Jun 1;13(3):191-211. doi: 10.2165/11595240-000000000-00000.

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