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系统性硬化症与系统性红斑狼疮重叠综合征患者自体干细胞移植后的微血管重塑:一例报告

Microvascular remodeling after autologous stem cell transplant in an overlap of systemic sclerosis and systemic lupus erythematosus: A case report.

作者信息

Mumtaz Sehreen, Balistreri Lisa M, Abril Andy, Ayala Ernesto, Butendieck Ronald R, Berianu Florentina

机构信息

Division of Rheumatology, Mayo Clinic School of Graduate Medical Education, Mayo Clinic College of Medicine and Science, Jacksonville, FL, USA.

Division of Rheumatology, Mayo Clinic, Jacksonville, FL, USA.

出版信息

SAGE Open Med Case Rep. 2024 Aug 22;12:2050313X241271811. doi: 10.1177/2050313X241271811. eCollection 2024.

DOI:10.1177/2050313X241271811
PMID:39185071
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11342424/
Abstract

Microvascular remodeling and capillary repopulation can occur after autologous hematopoietic stem cell transplant (HSCT) in patients with systemic sclerosis and systemic lupus erythematosus (SLE). We aim to report evidence for microvascular remodeling after autologous HSCT as observed by nailfold videocapillaroscopy (NVC). We describe a rare occurrence of features consistent with systemic sclerosis and SLE in a 33-year-old female with a complex clinical course refractory to conventional treatments, ultimately requiring autologous HSCT. We performed NVC before and after HSCT using optical video and light microscopy. At the microvascular level, morphologic changes in the capillary vascular bed were observed after HSCT. Pretransplant damage in capillary structure was noted as evidenced on NVC with architectural loss, ramifications, capillary drop, and decreased density. Posttransplant NVC revealed an increase in capillary density with evidence of microvascular remodeling. Further studies on the clinical use and impact of microvascular remodeling on disease progression are needed and looking into the application of NVC scoring to assess clinical response would be meaningful.

摘要

在系统性硬化症和系统性红斑狼疮(SLE)患者中,自体造血干细胞移植(HSCT)后可发生微血管重塑和毛细血管再填充。我们旨在报告通过甲襞视频毛细血管镜检查(NVC)观察到的自体HSCT后微血管重塑的证据。我们描述了一名33岁女性罕见地出现与系统性硬化症和SLE一致的特征,其临床病程复杂,对传统治疗无效,最终需要自体HSCT。我们在HSCT前后使用光学视频和光学显微镜进行了NVC检查。在微血管水平上,HSCT后观察到毛细血管血管床的形态学变化。NVC显示移植前毛细血管结构受损,表现为结构丧失、分支、毛细血管减少和密度降低。移植后NVC显示毛细血管密度增加,有微血管重塑的证据。需要进一步研究微血管重塑的临床应用及其对疾病进展的影响,研究NVC评分在评估临床反应中的应用将具有重要意义。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2bb9/11342424/9a2d571b6f3c/10.1177_2050313X241271811-fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2bb9/11342424/a30433a9e551/10.1177_2050313X241271811-fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2bb9/11342424/9a2d571b6f3c/10.1177_2050313X241271811-fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2bb9/11342424/a30433a9e551/10.1177_2050313X241271811-fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2bb9/11342424/9a2d571b6f3c/10.1177_2050313X241271811-fig2.jpg

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本文引用的文献

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Diagnostics (Basel). 2023 Jun 28;13(13):2204. doi: 10.3390/diagnostics13132204.
2
Correlation between Microvascular Damage and Internal Organ Involvement in Scleroderma: Focus on Lung Damage and Endothelial Dysfunction.硬皮病微血管损伤与内脏器官受累之间的相关性:聚焦于肺损伤与内皮功能障碍
Diagnostics (Basel). 2022 Dec 25;13(1):55. doi: 10.3390/diagnostics13010055.
3
Case report: A successful second autologous hematopoietic stem cell transplantation in refractory systemic sclerosis, with positive effect on skin involvement, pulmonary function and microcirculation.
病例报告:一例难治性系统性硬化症患者成功进行了第二次自体造血干细胞移植,对皮肤受累、肺功能和微循环均有积极影响。
Front Immunol. 2022 Nov 23;13:925776. doi: 10.3389/fimmu.2022.925776. eCollection 2022.
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Stem Cell Therapy in Lupus.狼疮的干细胞治疗
Rheumatol Immunol Res. 2022 Jul 6;3(2):61-68. doi: 10.2478/rir-2022-0011. eCollection 2022 Jun.
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Improvement of microangiopathy after haematopoietic stem cell transplantation in systemic sclerosis.系统性硬皮病患者造血干细胞移植后微血管病变的改善。
Clin Exp Rheumatol. 2022 Oct;40(10):1993-1998. doi: 10.55563/clinexprheumatol/5rjvkc. Epub 2022 Oct 17.
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Nailfold capillaroscopy in systemic sclerosis - state of the art: The evolving knowledge about capillaroscopic abnormalities in systemic sclerosis.系统性硬化症中的甲襞毛细血管镜检查——最新进展:关于系统性硬化症毛细血管镜异常的不断发展的知识。
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