Lin-lin Zhu, Xiu-he Lyu, Tian-tian Lei, Jin-lin Yang

1 Department of General Practice, General Practice Medical Center, West China Hospital, Sichuan University, Chengdu 610041, China

2 Department of Gastroenterology & Hepatology, West China Hospital, Sichuan University, Chengdu 610041, China

3 Sichuan University-Oxford University Huaxi Gastrointestinal Cancer Center, West China Hospital, Sichuan University, Chengdu 610041, Sichuan Province, China

Dear editor,

Boerhaave’s syndrome is a barogenic tear of the esophagus, typically at the gastroesophageal junction, caused by a sudden increase in intraluminal pressure in the distal esophagus.[1]In recent years, the number of Boerhaave’s syndrome cases has increased, and a growing proportion of clinicians have recognized this rare but life-threatening disease. Untimely diagnosed patients showed a significant association with high mortality, which was ＞56% after 24 h and 75%-89% after 48 h.[2]Emergency physicians play a critical role in the early rapid detection and diagnosis of Boerhaave’s syndrome by arranging reasonable examinations and developing a multidisciplinary team. Herein, we report a case from China of a patient who developed spontaneous esophageal rupture during diagnostic colonoscopy. We aim to determine the optimal management strategies for Boerhaave’s syndrome from an emergency physician’s perspective.

CASE

A 48-year-old male was admitted to the emergency department complaining of abdominal distension and shortness of breath after undergoing a diagnostic colonoscopy for approximately 2 h. The patient was healthy, had no specific comorbidities, and had no previous medical or family history of significant abnormalities. Approximately 10 h before admission, he vomited pure gastric juice several times during the process of bowel preparation. During the examination, the patient developed cough and presented with hypoxemia (oxygen saturation [SaO2] 75%-85%). After resuscitation, he experienced upper abdominal distension accompanied by shortness of breath. Immediate arterial blood gas analysis indicated that the pH was 7.37, partial pressure of carbon dioxide (PCO2) 34.8 mmHg (1 mmHg=0.133 kPa), partial pressure of oxygen (PO2) 51.9 mmHg, and SaO287%.

Physical examination showed a high respiratory rate (40 breaths/min) with orthopnea, right deviation of the trachea, decreased breath sounds of the left upper lung, and absence of breath sound of the left lower lung. Pleural fluid analysis showed that red blood cells were 20,000×106/L, nucleated cells were 15,240×106/L, and pyocytes were positive. Pleural effusion biochemistry showed that albumin was 19.7 g/L, glucose 0.30 mmol/L, and lactate dehydrogenase 771 U/L. Enhanced computed tomography (CT) of the chest and abdomen suggested massive left-sided pleural effusion, mediastinal pneumatosis, and suspected perforation of the esophagus (Figure 1A). No blue liquid was discovered in the chest drainage tube after administering methylene blue twice during hospitalization. Three days after admission, enhanced CT of the chest showed contrast agent entering the chest from the esophagus, and an esophageal-thoracic fistula was considered (Figure 1B).

After continuous left chest drainage, oxygen inhalation by mask, anti-infection and nutritional support, the patient’s symptoms were partially alleviated with stable breathing in the supine position. Then, videoassisted thoracoscopic surgery (VATS) was performed, including stripping of the left pyothorax fiberboard, drainage of the pyothorax, and cautery division of pleural adhesions. This surgery revealed wide extensive pleural adhesion, pus accumulation and compartmentalization in the left pleural cavity. A 4.0 cm×1.0 cm perforation was located above the esophageal hiatus and in the medial part of the thoracic aorta. We did not perform primary repair to the esophagus but instead only drainage to the thoracic cavity.

After surgery, the patient gradually improved, and chest CT showed a small amount of effusion and a thickened pleural membrane approximately 10 d after surgery (Figure 1C). The patient recovered relatively well and was discharged 3 weeks after admission.

Figure 1. Computed tomography (CT) images of the patient at different stages of disease. A: CT shows left pleural effusion with an unclear esophageal boundary. B: CT shows that the contrast agent enters the chest from the esophagus; red arrows represents that the contrast agent enters the chest from the esophagus; C: CT shows a small amount of effusion and a thickened pleural membrane; red arrows represents a thickened pleural membrane.

DISCUSSION

Our patient was the case from China of a patient who developed Boerhaave’s syndrome during diagnostic colonoscopy. Only one study has reported a similar case until now;[3]the patient underwent a low esophageal perforation after diagnostic colonoscopy, possibly due to bowel-cleansing preparation. We considered that intense cough was the root cause of the condition in our patient. Colonoscopy may have increased the patient’s i ntrathoracic pressure due to stimulation of the vagus nerve, which caused the patient to cough. This cough thereby exacerbated the condition and ultimately caused perforation.[4]In fact, almost all current reports of similar patients were in the emergency department, and our case is not an exception. Therefore, we want to address some insights from the emergency physician’s perspective.

First, the patient’s symptoms and medical history before onset play a pivotal role in identifying this rare disease. We suggest that vomiting and chest pain be considered red flags because they manifest in at least 50% of B oerhaave’s syndrome cases.[5]In addition, forceful emesis or coughing, undergoing gastrointestinal endoscopy,[6]eating heavy meals, excessive alcohol consumption, having epileptic seizures, and asthma attacks could also cause a sharp increase in intraluminal pressure, thus leading to esophageal rupture. Accordingly, there were higher requirements on the awareness of Boerhaave’s syndrome for emergency physicians.

Second, regarding the preferred diagnostic method, CT scans, esophagograms or endoscopy can provide diagnostic information. CT and esophagogram by oral meglumine diatrizoate can be considered for patients who are not suitable for invasive examinations. However, small esophageal perforations could be overlooked on CT scans because the swollen esophageal wall may close the fistula orifice. Endoscopy has high sensitivity and specificity in the diagnosis of esophageal rupture, and it allows for the direct visualization of the specific perforation site and the size of the defect. Two important prerequisites allowing esophagogastroscopy were a stable patient condition without septic complications and adequate drainage of septic contamination from the chest.[7]Our patient was unable to undergo esophagogastroscopy due to unstable vital signs. Esophageal perforation was finally confirmed by oral meglumine diatrizoate esophagogram 80 h after onset.

Therefore, it is crucial for emergency physicians to assemble a multidisciplinary team (MDT) to develop urgent and individualized regimes. Gastroenterologists, radiologists, surgeons, and emergency physicians should discuss the most accurate examination technique once esophageal rupture is suspected.

Third, MDTs should make further decisions regarding individualized treatment strategies. Treatment options include conservative, endoscopic or operative modalities. Common endoscopic interventions include self-expanding stents[7]and endoluminal vacuum therapy.[8]Endoscopic negative pressure therapy (ENPT) is a novel method to treat Boerhaave’s syndrome.[9]Open-pore polyurethane foam drains with long drainage elements were used to cover the long perforation defect, and the contaminated wound secretion and liquid digestive tract were extracted under intraluminal endoscopic negative pressure.[10]ENPT combined with surgery, such as laparotomy with abdominal lavage, has also been suggested in a complementary manner,[9]thus directly contributing to wound healing. Customized patient treatment depends on the patients’ condition, lesion extension and time from symptom onset.

Finally, follow-up is important to recognize Boerhaave’s syndrome, especially for emergency physicians. Most patients were discharged from the gastroenterology department or thoracic surgery, and emergency physicians should address specific concerns regarding treatment options and patient outcomes to increase their level of experience.

CONCLUSION

Emergency physicians play an irreplaceable role in all aspects from medical history collection to follow-up. It is necessary to investigate this rare disease from an emergency physician’s perspective.

Funding:This work was supported by the Sichuan Science and Technology Program (22GJHZ0177).

Ethical approval:Written consent for publication was obtained from the patient’s family.

Confl icts of interest:They have no confl icts of interest.

Contributors:LLZ reviewed the literature and wrote the manuscript draft. TTL and XHL collected the patient’s data. JLY revised the manuscript for important intellectual content. All authors approved the version to be submitted.