1 型神经纤维瘤病患者胸内动脉瘤破裂的血管内治疗:病例报告。

IF 0.7 Q4 SURGERY Surgical Case Reports Pub Date : 2024-08-30 DOI:10.1186/s40792-024-02002-9
Ryoma Oda, Daisuke Endo, Takeshi Udagawa, Shingo Okada, Ryohei Kuwatsuru, Minoru Tabata
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引用次数: 0

摘要

背景:胸内动脉瘤(ITAA)是一种极为罕见的疾病,约三分之二的报告病例为先天性假性动脉瘤。其余病例由各种原因引起,包括血管炎、结缔组织病和神经纤维瘤病 1 型(NF-1)。NF-1 是一种常染色体显性遗传疾病,临床表现各不相同,偶尔会出现危及生命的血管并发症。尽管NF-1患者可能会出现各种血管异常,但ITAA破裂的病例却鲜有报道,已发表的病例仅有7例:一名 32 岁的 NF-1 男性患者因持续性左背部和上臂疼痛三天而就诊。初步胸片检查显示左侧胸腔积液,左肺顶有不透明物。计算机断层扫描显示左上纵隔有肿块,起初怀疑是肿瘤。随后的对比增强计算机断层扫描显示,肿块是锁骨下动脉瘤。为了制定手术计划,该患者接受了1毫米切片的详细对比增强计算机断层扫描,结果显示肿块为左侧ITAA,并伴有破裂。考虑到再次破裂的风险,患者接受了急诊血管造影术,结果证实左侧ITAA破裂,但没有外渗。在近端和远端使用多个微线圈成功治疗了ITAA。患者恢复顺利,术后第四天出院:本病例强调了在NF-1患者出现胸腔积液时考虑血管病变的重要性。结论:本病例强调了NF-1患者出现胸腔积液时考虑血管病变的重要性,同时也强调了诊断ITAA的挑战以及薄层对比增强计算机断层扫描和血管内治疗的有效性。
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Endovascular treatment of contained ruptured internal thoracic artery aneurysm mimicking a tumor in a patient with neurofibromatosis type 1: a case report.

Background: An internal thoracic artery aneurysm (ITAA) is an exceedingly rare condition, with approximately two-thirds of reported cases being iatrogenic pseudoaneurysms. The remainder are attributed to various causes, including vasculitis, connective tissue disease, and neurofibromatosis type 1 (NF-1). NF-1 is an autosomal dominant disorder characterized by distinct clinical manifestations that occasionally include life-threatening vascular complications. Although NF-1 patients may develop various vascular abnormalities, ruptured ITAA is rarely reported, with only seven published cases.

Case presentation: A 32-year-old man with NF-1 consulted for a three-day history of persistent left back and upper arm pain. Initial chest radiography indicated left pleural effusion and an opacity at the left lung apex. Computed tomography scan revealed a mass in the left upper mediastinum that was initially suspected to be a tumor. Subsequent contrast-enhanced computed tomography revealed the mass to be a subclavian artery aneurysm. Detailed contrast-enhanced computed tomography with 1-mm slices was performed for surgical planning, identifying the mass as a left ITAA with contained rupture. Given the risk of re-rupture, emergency angiography was performed, which confirmed rupture of the left ITAA without extravasation. The ITAA was successfully treated with multiple microcoils at the proximal and distal ends. The patient had an uneventful recovery and was discharged on the fourth postoperative day.

Conclusions: This case highlights the importance of considering vascular lesions in NF-1 patients who present with pleural effusion. It also emphasizes the challenges in diagnosing ITAA and the effectiveness of thin-slice contrast-enhanced computed tomography scans and endovascular treatment.

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