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继发于巨大奇静脉血管瘤的难治性乳糜胸:挑战性病例报告的定制多模式治疗。

Refractory Chylothorax Secondary to Sizeable Azygos Vein Hemangioma: Tailored Multimodal Treatment of a Challenging Case Report.

机构信息

Division of Thoracic Surgery, Azienda di Rilievo Nazionale ed Alta Specializzazione "G. Brotzu", 09121 Cagliari, Italy.

Neuroradiology and Vascular Radiology Unit, Azienda di Rilievo Nazionale ed Alta Specializzazione "G. Brotzu", 09121 Cagliari, Italy.

出版信息

Medicina (Kaunas). 2022 Dec 31;59(1):91. doi: 10.3390/medicina59010091.

Abstract

BACKGROUND

Mediastinal hemangiomas are rare, and their etiology remains unclear. Most patients affected have no pathognomonic clinical symptoms, and the diagnosis is often incidental. Due to the paucity of the available literature regarding the management of this disease, the choice and timing of treatment remains controversial.

CASE PRESENTATION

Herein, we report the case of a hemangioma of the azygos vein arch in a 66-year-old woman who presented with dyspnea, chest discomfort, dysphagia, and weight loss. A simultaneous right chylothorax refractory to conservative management was found. A CT-guided biopsy of the mass was performed, and it confirmed the vascular nature of the lesion. Therefore, the patient underwent an angiography followed by endo-vascular embolization. Three days later, thoracoscopic surgical resection of the mass and the repair of the chyle leakage were performed safely. The patient was discharged uneventfully on postoperative day seven, with complete resolution of all the presenting symptoms.

CONCLUSIONS

Treatment of symptomatic mediastinal hemangiomas could be mandatory, but a thorough multidisciplinary approach to these rare malformations is essential. Despite the risk of intraoperative bleeding, selective endovascular embolization followed by thoracoscopic surgery allowed for a complete and safe resection with a good outcome.

摘要

背景

纵隔血管瘤罕见,其病因仍不清楚。大多数受影响的患者没有明显的临床症状,诊断通常是偶然的。由于关于这种疾病的管理的文献有限,治疗的选择和时机仍然存在争议。

病例介绍

在此,我们报告了一位 66 岁女性的奇静脉弓血管瘤病例,她表现为呼吸困难、胸痛、吞咽困难和体重减轻。同时发现右侧乳糜胸难治性。对肿块进行了 CT 引导下活检,证实了病变的血管性质。因此,患者进行了血管造影检查,随后进行了血管内栓塞。3 天后,安全地进行了胸腔镜下肿块切除术和乳糜漏修复术。患者术后第 7 天顺利出院,所有症状完全缓解。

结论

治疗有症状的纵隔血管瘤可能是强制性的,但对这些罕见畸形进行彻底的多学科方法是必要的。尽管存在术中出血的风险,但选择性血管内栓塞后行胸腔镜手术可安全完整地切除,预后良好。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b19d/9863118/795a366a4f9d/medicina-59-00091-g001.jpg

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