Boerhaave’s Syndrome: Delayed Management Using Over-the-Scope Clip

Background

Boerhaave’s syndrome is spontaneous perforation of the esophagus; most commonly the distal part, as a result of forceful emesis. The classical triad of sub-xiphoid retrosternal pain, vomiting, and subcutaneous emphysema is present in a small number of patients, posing a formidable diagnostic challenge [1]. The management option depends on the time of perforation. Early presentation (within 24 hours) is commonly managed surgically, but delayed presentation is managed conservatively, especially if the perforation is confined and the clinical condition of the patient is stable. However, delayed presentation and treatment is associated with inferior outcome [1]. In recent years, endoscopic options are gaining popularity in the management of Boerhaave’s syndrome. Such options include endoscopic stenting and over-the-scope clipping (OTSC) of the perforation [2,3]. We report here a case of successful management by the OTSC system of a patient with Boerhaave’s syndrome who presented more than 9-days after the perforation.

Case Report

OUTCOME AND FOLLOW-UP:

The patient’s condition improved, and he was discharged home on day 16 after admission. The patient was seen in the out-patient clinic 14 days after discharge. He was well and tolerating full diet with no other complications. A chest x-ray was done at that time was normal (Figure 6). He remained well at 6-month follow-up.

Discussion

Spontaneous esophageal rupture (Boerhaave’s syndrome) is rarely encountered and hence its diagnosis is either missed or delayed leading to a high mortality rate that approaches 60% if time to theatre is delayed beyond 48 hours. However, the overall mortality in cases managed early, within 24 hours of presentation, is much lower at 20% [4]. In addition to the delay in diagnosis and treatment, another major prognostic factor in determining mortality is the site of the injury.

It is recommended that all septic patients with esophageal perforation should be treated surgically, and conservative treatment is reserved for patients presenting early without sepsis. Conservative treatment is also advocated for those with delayed presentations but without sepsis. The recovery of such patients can be hastened by minimally invasive endoscopic means [2].

The hospital stay of our patient was 16 days; this was much shorter than that reported by others for delayed Boerhaave’s syndrome treated by OTSC [5]. There is no doubt that the combined non-operative interventional radiology drainage of septic collection and the minimally invasive OTSC greatly contributed to the rapid recovery and short hospital stay. This combined management strategy emphasizes the importance of a multi-disciplinary team approach for the management of this condition to minimize morbidity and mortality, and to achieve good outcomes.

In recent years, an increasing number of patients with esophageal perforations are being managed by non-operative endoscopic measures such as placement of covered self-expanding stents, over-the-scope clipping, or both a clip and a stent [6]. A covered self-expanding stent can achieve sealing of a leak in up to 50% of cases, and placement of a second stent in failed cases is not usually successful in sealing the leak [1]. The OTSC system is a clipping method for mechanical compression of tissues in the gastrointestinal tract. It was first designed for the management of bleeding, perforation, and fistula [7], and hence, OTSC can be appropriately employed in the treatment of esophageal perforation [3,5,8]. It has been demonstrated that standard clips might not be as effective as OTSC as a primary therapy in sealing gastrointestinal bleeding [9]. In comparison to the conventional clips, OTSC has a greater compressive force and is more effective in achieving superior grip of fibrotic tissue, as in this case of delayed perforation. Moreover, OTSC offers easy application and maneuvering in tight anatomical positions. However, there is no clinical study to date that compares OTSC versus conventional clips, except one prospective randomized study that confirmed superiority of OTSC over standard methods used to control recurrent ulcer bleeding (not perforation) in term of technical and clinical success [10].

Our case presented late (9 days after perforation) to our facility and the size of the perforation was 1.5 cm. Due to the site of the perforation, OTSC rather than stenting was applied with success. However, the closure could not be achieved by one clip and therefore, a second clip was applied. The residual thoracic sepsis was treated by tube drainage and later by pleurodesis after clearance of sepsis. A review of the literature for cases of delayed Boerhaave’s syndrome treated by OTSC revealed 3 similar cases. The first successful endoscopic use of the OTSC in a delayed presentation (7 days after admission) of Boerhaave’s syndrome with mediastinitis was reported in 2013 by Ramhamadany et al. in a case of a 69-year-old patient. During endoscopy, the defect was visualized and successfully closed using OTSC, which resulted in a favorable outcome [3]. Bona et al. reported a case of a 36-year-old male patient referred with a delayed diagnosis of Boerhaave’s syndrome (12 days), which was successfully treated by OTSC application followed by lung decortication [5]. Kircheva and Vigneswaran reported a case of a 53-year-old male patient who had failed OTSC followed by endoscopic stenting after failure of OTSC to seal the leak. Despite stenting, the leak persisted necessitating a primary surgical repair 14 days after perforation [11]. This case confirms that combining OTSC and stenting does not guarantee success and does not avert the need for surgery in some cases. It also indicates that successful primary repair can be done in delayed perforations (as late as 2 weeks after the diagnosis) and after failure of the endoscopic management [11].

Endoscopic stenting alone is another viable option even in delayed cases [12]. Aloreidi et al. reported effective endoscopic stent treatment in 6 patients (4 males and 2 females) with Boerhaave’s syndrome. They attributed satisfactory results in patients presenting with sepsis to urgent interventional radiology-guided fluid drainage [12]. This again highlights the importance of a multidisciplinary team approach in the management of this condition to secure good and favorable outcomes. Other minimally invasive mean of treating delayed esophageal rupture is by T-tube drainage using video-assisted thoracoscopic surgery (stay 46 days) [13].

Conclusions

OTSC of esophageal perforation in Boerhaave’s syndrome presenting late is safe and effective. Moreover, OTSC hastens recovery and shortens the hospital stay in cases suitable for conservative treatment.