低温等离子射频消融联合多模式支气管镜介入治疗气管支气管淀粉样变性所致气道狭窄：一例病例报告及文献复习

文献信息

中文摘要

背景： 气管支气管淀粉样变性（TBA）是一种罕见的气道疾病，其特征是淀粉样蛋白异常沉积于气管支气管树的黏膜下层和肌层。临床表现缺乏特异性，常被误诊为慢性阻塞性肺疾病、支气管哮喘或气道恶性肿瘤。目前尚无标准化的诊断和治疗指南，支气管镜介入已成为解除气道阻塞的首选方法。方法：我们报告一名56岁男性TBA患者，表现为12年反复咳嗽、咳痰和呼吸困难病史。胸部计算机断层扫描（CT）显示弥漫性支气管壁增厚伴钙化及不规则管腔狭窄，右中叶和下叶可见阻塞性肺炎和肺不张。支气管镜检查显示弥漫性黏膜增厚伴结节状增生和明显管腔狭窄。刚果红染色阳性，偏振光下呈苹果绿双折射，证实为局限性TBA。患者在12个月内接受了三个阶段的多种模式支气管镜介入治疗——包括球囊扩张、钳夹清创、二极管激光消融、低温等离子射频消融（LTP-RFA）和冷冻治疗——以治疗原发性、复发性和残留性气道狭窄。结果：经过系列治疗后，患者症状明显改善，内镜和影像学均有改善。结论：本病例是LTP-RFA应用于TBA的少数报告之一。我们复习了近期关于TBA治疗方式和结局的文献，为个体化TBA管理提供参考。

英文摘要

Tracheobronchial amyloidosis (TBA) is a rare airway disorder characterized by abnormal deposition of amyloid protein in the submucosa and muscular layer of the tracheobronchial tree. Clinical manifestations are nonspecific, and the disease is frequently misdiagnosed as chronic obstructive pulmonary disease, bronchial asthma, or airway malignancy. No standardized diagnostic and treatment guidelines exist, and bronchoscopic interventions have become the preferred approach for relieving airway obstruction. We report a 56-year-old man with TBA presenting with a 12-year history of recurrent cough, sputum production, and dyspnea. Chest computed tomography (CT) revealed diffuse bronchial wall thickening with calcification and irregular luminal narrowing, with obstructive pneumonia and atelectasis in the right middle and lower lobes. Bronchoscopy demonstrated diffuse mucosal thickening with nodular hyperplasia and marked luminal stenosis. Congo red staining was positive with apple-green birefringence under polarized light, confirming localized TBA. The patient underwent three stages of multimodal bronchoscopic intervention over 12 months-comprising balloon dilatation, forceps debridement, diode laser ablation, low-temperature plasma radiofrequency ablation (LTP-RFA), and cryotherapy-to treat the primary, recurrent, and residual airway stenosis. Following serial treatments, the patient's symptoms improved significantly, with endoscopic and radiographic improvement. This case represents one of the few reports of LTP-RFA application in TBA. We review the recent literature on TBA treatment modalities and outcomes, providing a reference for individualized TBA management.