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特纳综合征合并肺动静脉畸形:一例报告

Turner syndrome with pulmonary arteriovenous malformation: a case report.

作者信息

Guo Huibin, Chen Hongqiao, Chen Sihao, Tang Shilong

机构信息

Department of Radiology, University-Town Hospital of Chongqing Medical University, Chongqing, China.

Department of Radiology, Children's Hospital of Chongqing Medical University, National Clinical Research Center for Child Health and Disorders, Ministry of Education Key Laboratory of Child Development and Disorders, Chongqing Key Laboratory of Pediatrics, Chongqing, China.

出版信息

Front Cardiovasc Med. 2025 Aug 28;12:1603250. doi: 10.3389/fcvm.2025.1603250. eCollection 2025.

Abstract

Turner syndrome (TS) is the most common sex chromosome abnormality disorder, caused by complete or partial absence of the X chromosome, its clinical manifestations primarily include short stature, gonadal dysgenesis, and characteristic cardiovascular malformations, pediatric cardiologists pay particular attention to coarctation of the aorta (CoA), which occurs in 15%-30% of TS patients and represents a life-threatening condition requiring prioritized screening during the neonatal and childhood periods (1- 3). Furthermore, due to lymphatic system developmental abnormalities, TS patients also face elevated risks of aortic root dilation, bicuspid aortic valve, and vascular structural anomalies (3). Pulmonary arteriovenous malformation (PAVM) is a rare pulmonary vascular anomaly, with an estimated prevalence of approximately 1 in 50,000 in the general population (1-3). Although the exact prevalence of PAVM in TS patients remains unclear, case series suggest a significantly elevated risk compared to the general population (estimated risk ratio: 5- to 10-fold), this association may be attributed to defective vascular elastic fiber development and dysregulated angiogenic signaling pathways in TS patients (4, 5). Here, we report the first documented case of TS complicated by PAVM, aiming to enhance clinicians' awareness of this rare comorbidity and provide evidence-based diagnostic and therapeutic recommendations.

摘要

特纳综合征(TS)是最常见的性染色体异常疾病,由X染色体完全或部分缺失引起,其临床表现主要包括身材矮小、性腺发育不全以及特征性心血管畸形,儿科心脏病专家特别关注主动脉缩窄(CoA),该病在15% - 30%的TS患者中出现,是一种危及生命的疾病,需要在新生儿期和儿童期进行优先筛查(1 - 3)。此外,由于淋巴系统发育异常,TS患者还面临主动脉根部扩张、二叶式主动脉瓣和血管结构异常的风险增加(3)。肺动静脉畸形(PAVM)是一种罕见的肺血管异常,在普通人群中的估计患病率约为万分之一(1 - 3)。虽然TS患者中PAVM的确切患病率尚不清楚,但病例系列研究表明,与普通人群相比,其风险显著升高(估计风险比:5至10倍),这种关联可能归因于TS患者血管弹性纤维发育缺陷和血管生成信号通路失调(4, 5)。在此,我们报告首例记录在案的TS合并PAVM病例,旨在提高临床医生对这种罕见合并症的认识,并提供基于证据的诊断和治疗建议。

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