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1例伴有血管性血友病因子缺乏和多发副脾的Klippel Trenaunay综合征罕见病例:病例报告及文献简要综述

A Rare Case of Klippel Trenaunay Syndrome with Von Willebrand Factor Deficiency and Multiple Accessory Spleens: A Case Report and Brief Literature Review.

作者信息

Falahati Vahid, Fallahi Mahsa, Shahriarpour Mona, Ghasemi Ali, Ghaffari Kazem

机构信息

Department of Pediatric Hematology and Oncology, Faculty of Medicine, Arak University of Medical Sciences, Arak, Iran.

Department of Dermatology, Faculty of Medicine, Arak University of Medical Sciences, Arak, Iran.

出版信息

Adv Biomed Res. 2024 Feb 26;13:20. doi: 10.4103/abr.abr_232_23. eCollection 2024.

DOI:10.4103/abr.abr_232_23
PMID:38525403
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10958732/
Abstract

Klippel Trenaunay Syndrome (KTS) is an uncommon inherited syndrome identified by venous varicosities and capillary abnormalities. von Willebrand Disease is the most common inherited hemorrhage disturbance in humans, leading to insufficiency in von Willebrand Factor, which is a complex multimeric protein with two functions: it forms a bridge between the platelets and injured vascular areas and it attaches factor VIII and stabilizes it. We present a 13-year-old son with a typical clinical manifestation of KTS, including "port-wine stains" as capillary malformation, venous malformation, and hypertrophy of the left lower extremity, who also suffers from von Willebrand Disease type 3. He has been suffering from these two rare conditions since birth. The occurrence of KTS with von Willebrand Factor deficiency in a patient has so far not been reported, which may propose a mutation in the putative common regulatory gene that caused this uncommon phenotype.

摘要

克-特综合征(KTS)是一种罕见的遗传性综合征,其特征为静脉静脉曲张和毛细血管异常。血管性血友病是人类最常见的遗传性出血障碍,会导致血管性血友病因子不足,该因子是一种具有两种功能的复杂多聚体蛋白:它在血小板和受损血管区域之间形成桥梁,并附着凝血因子VIII并使其稳定。我们报告一名13岁男性,具有典型的KTS临床表现,包括作为毛细血管畸形的“葡萄酒色斑”、静脉畸形和左下肢肥大,他还患有3型血管性血友病。他自出生以来就患有这两种罕见疾病。迄今为止,尚未报道过患者同时患有KTS和血管性血友病因子缺乏症的情况,这可能提示在假定的共同调控基因中发生了突变,导致了这种罕见的表型。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8554/10958732/477b2f0abffa/ABR-13-20-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8554/10958732/477b2f0abffa/ABR-13-20-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8554/10958732/477b2f0abffa/ABR-13-20-g001.jpg

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本文引用的文献

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Status of major hemostatic components in the setting of COVID-19: the effect on endothelium, platelets, coagulation factors, fibrinolytic system, and complement.新型冠状病毒肺炎背景下主要止血成分的状态:对血管内皮、血小板、凝血因子、纤维蛋白溶解系统和补体的影响。
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Klippel-Trenaunay syndrome belongs to the PIK3CA-related overgrowth spectrum (PROS).克-特综合征属于PIK3CA相关过度生长谱系(PROS)。
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Blood. 2013 Mar 21;121(12):2336-9. doi: 10.1182/blood-2012-09-455089. Epub 2013 Jan 24.