脂质坏死出现在用局部鲁索利替尼治疗的葡萄酒染色。

IF 1.1 Q2 MEDICINE, GENERAL & INTERNAL
Neena Edupuganti, Jay Nguyen, Fahad Siddiqui, Victoria Dukharan, Rajiv Nathoo
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引用次数: 0

摘要

我们提出了一个独特的病例脂质坏死症(NL)产生在波特酒染色(PWS),这,据我们所知,以前没有报道过。NL是一种罕见的肉芽肿性疾病,常与糖尿病相关,以慢性炎症和微血管功能障碍为特征。PWS是一种先天性血管畸形,由内皮细胞分化缺陷和毛细血管畸形引起。这些疾病的共存表明它们的病理生理机制存在潜在的联系,包括炎症、内皮功能障碍和共享的信号通路,如丝裂原活化蛋白激酶(MAPK)、磷酸肌肽3-激酶(PI3K)和肿瘤坏死因子(TNF)- α。在包括氯倍他索、他克莫司和脉冲染料激光在内的常规治疗失败后,患者的NL病变在局部使用ruxolitinib(一种Janus激酶(JAK)抑制剂)后得到改善。据我们所知,这是第一例局部鲁索利替尼单药治疗成功的NL病例。本病例强调了JAK抑制剂在肉芽肿疾病管理中的新作用,并提出了关于炎症和血管过程之间共享的病理生理机制的有趣问题。进一步研究细胞因子失调、免疫反应和针对这些重叠病理的靶向治疗可以提供更有效的治疗策略并改善患者的预后。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Necrobiosis lipoidica arising in a port wine stain treated with topical ruxolitinib.

We present a unique case of necrobiosis lipoidica (NL) arising within a port-wine stain (PWS), which, to our knowledge, has not been previously reported. NL is a rare granulomatous disorder often associated with diabetes mellitus, characterized by chronic inflammation and microvascular dysfunction. PWS, a congenital vascular malformation, results from defective endothelial differentiation and capillary malformation. The coexistence of these conditions suggests a potential link in their pathophysiological mechanisms, including inflammation, endothelial dysfunction, and shared signaling pathways such as mitogen-activated protein kinase (MAPK), phosphoinositide 3-kinase (PI3K), and tumor necrosis factor (TNF)-alpha. Our patient's NL lesions demonstrated improvement with topical ruxolitinib, a Janus kinase (JAK) inhibitor, after failing conventional therapies, including clobetasol, tacrolimus, and pulsed dye laser. To our knowledge, this is the first reported case of NL successfully treated with topical ruxolitinib monotherapy. This case highlights the emerging role of JAK inhibitors in managing granulomatous disorders and raises intriguing questions about the shared pathophysiologic mechanisms between inflammatory and vascular processes. Further investigation into cytokine dysregulation, immune responses, and targeted therapies for these overlapping pathologies could inform more effective treatment strategies and improve patient outcomes.

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来源期刊
Journal of Osteopathic Medicine
Journal of Osteopathic Medicine Health Professions-Complementary and Manual Therapy
CiteScore
2.20
自引率
13.30%
发文量
118
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