Li Rina, Skeik Nedaa
Minneapolis Heart Institute Foundation, Minneapolis, MN.
Division of Vascular Medicine, Thrombophilia and Anticoagulation Clinic, Vein Center, Abbott Northwestern Hospital/Minneapolis Heart Institute, Minneapolis, MN.
J Vasc Surg Cases Innov Tech. 2024 Jan 17;10(2):101428. doi: 10.1016/j.jvscit.2024.101428. eCollection 2024 Apr.
Bannayan-Riley-Ruvalcaba syndrome (BRRS) is a congenital, autosomal-dominant disorder characterized by a triad of macrocephaly, lipomatosis, and pigmentation of the glans penis. The symptoms of this rare syndrome vary greatly and include multiple hamartomatous polyps, macrocephaly, increased birth weight, developmental delay, and intellectual disability. Vascular abnormalities, including arteriovenous malformations (AVMs), have rarely been reported as part of the vascular manifestations associated with BRRS. Congenital AVMs can rarely progress, resulting in limb- or life-threatening complications. We present the case of a young man with BRRS diagnosed in childhood and presenting with three AVMs involving the right upper extremity and chest. We also provide a brief literature summary of reported cases of BRRS with AVMs. Our paper highlights the importance of recognizing and understanding the vascular manifestations in patients with BRRS. Knowledge of the association between BRRS and AVMs is crucial for guiding patient diagnosis and management, optimizing treatment strategies, and improving overall patient outcomes.
班纳扬-莱利-鲁瓦尔卡瓦综合征(BRRS)是一种先天性常染色体显性疾病,其特征为巨头畸形、脂肪瘤病和阴茎头色素沉着三联征。这种罕见综合征的症状差异很大,包括多发性错构瘤性息肉、巨头畸形、出生体重增加、发育迟缓以及智力残疾。血管异常,包括动静脉畸形(AVM),作为与BRRS相关的血管表现的一部分,鲜有报道。先天性AVM很少进展,导致肢体或危及生命的并发症。我们报告一例童年时诊断为BRRS的年轻男性病例,其出现累及右上臂和胸部的三处AVM。我们还简要总结了已报道的伴有AVM的BRRS病例的文献。我们的论文强调了识别和理解BRRS患者血管表现的重要性。了解BRRS与AVM之间的关联对于指导患者诊断和管理、优化治疗策略以及改善患者总体预后至关重要。