Zeyara Adam, Jeremiasen Martin, Åkesson Oscar, Falkenback Dan, Hermansson Michael, Johansson Jan
Department of Clinical Sciences in Surgery, Lund University, Lund, Sweden.
Department of Surgery, Ystad Hospital, Ystad, Sweden.
J Med Case Rep. 2021 May 2;15(1):223. doi: 10.1186/s13256-021-02779-5.
Effort rupture of the esophagus or Boerhaave's syndrome is a rare entity, and prognosis is largely dependent on early diagnosis and treatment. Recurrent effort ruptures are very rare, only reported in a few case reports in English literature. We present a case with a third time effort rupture, and to the best of our knowledge there are no such previous publications. Furthermore, the presented case is also distinct because each episode was treated by different methods, reflecting the pathophysiology of recurrent disease as well as the last decade's advancements in the management of esophageal perforations in our clinic and globally.
The patient is a 60-year-old White male, suffering from alcohol abuse, mild reflux esophagitis, and a history of effort esophageal ruptures on two previous occasions. He was now admitted to our ward once again because of a third bout of Boerhaave's syndrome. The first time, 10 years ago, he was managed by thoracotomy and laparotomy with primary repair, and the second time, 5 years ago, by transhiatal mediastinal drainage through a laparotomy and endoscopic stent placement. Now he was successfully managed by endovascular vacuum-assisted closure therapy alone.
Recurrent cases of Boerhaave's syndrome are very rare, and treatment must be tailored individually. The basic rationale is, however, no different from primary disease: (1) early diagnosis, (2) adequate drainage of extraesophageal contamination, and (3) restoration of esophageal integrity. Recurrent disease is usually contained and exceptionally suitable for primary endoscopic treatment. To cover the full panorama and difficult nature of complex esophageal disease, endoscopic modalities such as stent placement and endovascular vacuum-assisted closure, as well as the capacity for prompt extensive surgical interventions such as esophagectomy, should be readily accessible within every modern esophageal center.
食管自发性破裂或Boerhaave综合征是一种罕见疾病，其预后很大程度上取决于早期诊断和治疗。复发性食管自发性破裂极为罕见，英文文献中仅有少数病例报告。我们报告一例第三次发生食管自发性破裂的病例，据我们所知，此前尚无此类报道。此外，该病例也很独特，因为每次发作的治疗方法不同，这既反映了复发性疾病的病理生理学，也体现了过去十年我们科室及全球范围内食管穿孔治疗的进展。
患者为一名60岁白人男性，有酗酒史、轻度反流性食管炎，既往有两次食管自发性破裂病史。因第三次发作Boerhaave综合征再次入住我们病房。第一次发作是在10年前，通过开胸和开腹手术进行一期修复；第二次发作是在5年前，通过开腹经裂孔纵隔引流及内镜下放置支架治疗。此次仅通过血管腔内负压封闭疗法成功治愈。
Boerhaave综合征复发病例非常罕见，治疗必须个体化。然而，基本原则与原发性疾病并无不同：（１）早期诊断；（２）充分引流食管外污染；（３）恢复食管完整性。复发性疾病通常可以控制，特别适合内镜下的初始治疗。为全面应对复杂食管疾病的全貌和困难特性，每个现代食管中心都应具备诸如支架置入、血管腔内负压封闭等内镜治疗手段，以及诸如食管切除术等及时进行广泛手术干预的能力。
