硼替佐米成功治疗缺血性非阻塞性冠状动脉疾病引发的难治性冷球蛋白性血管炎。

IF 1 Q4 UROLOGY & NEPHROLOGY
Yui Ohta, Takaaki Tsuchiya, Masatoshi Oka, Moriaki Tachibana, Yoshitaka Kondo, Kaoruko Fukushima, Shiho Matsuno, Noriko Yamanaka, Noriyuki Suzuki, Akiko Komatsu, Hirofumi Rokutan, Wako Yumura, Tomio Arai, Akihito Ishigami, Mitsuyo Itabashi, Takashi Takei
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引用次数: 0

摘要

I型和混合型冷球蛋白性血管炎在病理生理、临床表现和治疗反应上有所不同。我们报告一例难治性冷球蛋白性血管炎诊断缺血性非阻塞性冠状动脉疾病(INOCA)。患者表现为呼吸困难,以及缺血性肠炎、紫癜和肾衰竭引起的腹痛,需要透析。尽管患者存在IgG λ型未确定意义单克隆γ病(MGUS)和丙型肝炎病毒阴性,但冷球蛋白分析和强烈的肾小球IgM沉积提示类风湿因子(RF)活性的存在和IgM参与的可能性。病情被诊断为混合性冷球蛋白血症,并进行了各种免疫调节治疗,包括甲基强龙、利妥昔单抗和血浆分离,但未达到冷球蛋白阴性。然而,使用硼替佐米和地塞米松治疗最终导致冷球蛋白阴性和临床改善,尽管患者没有停止透析,导致冷球蛋白血症性血管炎缓解。本病例提示,硼替佐米,一种蛋白酶体抑制剂,可能是一种治疗难治性冷球蛋白性血管炎的有希望的方法。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Successful treatment with bortezomib for refractory cryoglobulinemic vasculitis triggered by ischemic non-obstructive coronary artery disease.

Type I and mixed cryoglobulinemic vasculitis differ in pathophysiology, clinical presentation, and therapeutic response. We report a case of refractory cryoglobulinemic vasculitis diagnosed following ischemic non-obstructive coronary artery disease (INOCA). The patient presented with dyspnea, as well as abdominal pain due to ischemic enteritis, purpura, and renal failure requiring dialysis. Despite the patient's IgG λ-type monoclonal gammopathy of undetermined significance (MGUS) and negative hepatitis C virus, the presence of rheumatoid factor (RF) activity and the possibility of IgM involvement were suggested by cryoglobulin analysis and strong glomerular IgM deposition. The condition was diagnosed as mixed cryoglobulinemia, and various immunomodulatory treatments, including methylprednisolone, rituximab and plasmapheresis, were administered without achieving cryoglobulin negativity. However, treatment with bortezomib and dexamethasone ultimately led to cryoglobulin negativity and clinical improvement although the patient was not weaned off dialysis, resulting in remission of the cryoglobulinemic vasculitis. This case suggests that bortezomib, a proteasome inhibitor, may be a promising treatment for refractory cryoglobulinemic vasculitis.

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来源期刊
CEN Case Reports
CEN Case Reports UROLOGY & NEPHROLOGY-
CiteScore
1.90
自引率
0.00%
发文量
80
期刊介绍: Clinical and Experimental Nephrology (CEN) Case Reports is a peer-reviewed online-only journal, officially published biannually by the Japanese Society of Nephrology (JSN).  The journal publishes original case reports in nephrology and related areas.  The purpose of CEN Case Reports is to provide clinicians and researchers with a forum in which to disseminate their personal experience to a wide readership and to review interesting cases encountered by colleagues all over the world, from whom contributions are welcomed.
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