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[右上叶肺隔离症作为复发性自发性气胸的罕见病因]

[Right upper lobe pulmonary sequestration as a rare cause of recurrent spontaneous pneumothorax].

作者信息

Agafonov G M, Petrov A S, Atyukov M A, Novikova O V, Zemtsova I Yu, Dvorakovskaya I V, Yablonsky P K

机构信息

Saint-Petersburg State University, St. Petersburg, Russia.

Saint-Petersbursg City Clinical Hospital No. 2, St. Petersburg, Russia.

出版信息

Khirurgiia (Mosk). 2024(1):102-109. doi: 10.17116/hirurgia2024011102.

DOI:10.17116/hirurgia2024011102
PMID:38258696
Abstract

A 19-year-old patient after previous wedge resection of the right upper pulmonary lobe a year ago urgently admitted with recurrent right-sided spontaneous pneumothorax. According to standard management of spontaneous pneumothorax, we performed diagnostic thoracoscopy and drainage of the right pleural cavity with regular X-ray examinations. However, these measures were ineffective. The patient was scheduled for surgery, and we intraoperatively observed an unusual cause of pneumothorax. Thus, we present spontaneous pneumothorax following right upper lobe pulmonary sequestration. The uniqueness of this case is associated with unusual manifestation and non-standard localization of rare lesion. A few cases of pneumothorax in similar patients are described in the world literature. The key limiting factor in diagnosis of such defects (identification of aberrant vessel supplying abnormal lung parenchyma) is the lack of routine CT angiography in patients diagnosed with pneumothorax. That is why CT changes were interpreted as postoperative ones, and the true cause was established only during redo surgery. A thorough inspection of the pleural cavity and alertness regarding unusual appearance of the right upper pulmonary lobe made it possible to suggest a non-standard diagnosis, avoid complications (bleeding from afferent vessel) and perform adequate lung resection.

摘要

一名19岁患者,一年前曾接受右上肺叶楔形切除术,现因复发性右侧自发性气胸紧急入院。根据自发性气胸的标准治疗方法,我们进行了诊断性胸腔镜检查并对右侧胸腔进行引流,并定期进行X线检查。然而,这些措施均无效。该患者被安排接受手术,术中我们观察到气胸的一个不寻常病因。因此,我们报告了右上叶肺隔离症继发的自发性气胸。该病例的独特之处在于罕见病变的不寻常表现和非标准定位。世界文献中描述了少数类似患者发生气胸的病例。诊断此类缺陷(识别供应异常肺实质的异常血管)的关键限制因素是气胸患者缺乏常规CT血管造影检查。这就是为什么CT改变被解释为术后改变,而真正的病因仅在再次手术时才得以确定。对胸腔进行彻底检查并对右上肺叶的异常外观保持警惕,使得我们能够提出非标准诊断,避免并发症(来自传入血管的出血)并进行充分的肺切除术。

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