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CASE REPORT
Year : 2017  |  Volume : 7  |  Issue : 1  |  Page : 30-35

Instrumental esophageal perforation: A case series


1 Department of Thoracic Surgery, Rizgari General Hospital, Erbil, Iraq
2 Department of Thoracic Surgery, Sulaimaniyah Teaching Hospital and School of Medicine, Faculty of Medical Sciences, University of Sulaimaniyah, Iraq
3 Department of General Surgery, Sulaimaniyah Teaching Hospital, Sulaimaniyah, Iraq

Date of Web Publication4-Apr-2018

Correspondence Address:
Dr. Abdulsalam Y Taha
Department of Thoracic Surgery, Sulaimaniyah Teaching Hospital and School of Medicine, Faculty of Medical Sciences, University of Sulaimaniyah, Sulaimaniyah
Iraq
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ais.ais_29_17

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  Abstract 


Esophageal perforation is a rare but dreadful surgical emergency. The main etiology is iatrogenic trauma, mostly related to endoscopic instrumentation. The treatment is controversial; however, surgery remains the mainstay. In this article, 3 cases of instrumental perforation of esophagus are described. In all cases, diagnosis was based on clinical, radiographic, and endoscopic findings. The first case was a 4-year-old girl with a corrosive stricture of esophagus who received 8 sessions of dilatation over 1 year; the last session resulted in perforation. Unfortunately, she was not referred to thoracic surgery at once but kept in gastroenterology department for several days, and hence, empyema developed. Conservative treatment was employed first followed by elective gastric pull-up via laparotomy and left neck incision 3 months later. She regained normal swallowing of fluid and semisolid food but developed a chronic neck sinus that required few drainage operations. The second case was of a 54-year-old man with dysphagia and a normal esophagus who developed perforation during rigid esophagoscopy. The perforation was immediately detected and an urgent primary repair was performed via left thoracotomy. The third case was of a 24-year-old man with persistent achalasia cardia despite 2 trans-abdominal esophagomyotomies. Perforation occurred during second session of pneumatic balloon dilatation but was directly referred to a thoracic surgeon who performed an urgent esophagectomy. All patients survived. We conclude that a good outcome of this potentially fatal condition is possible with early diagnosis and intervention provided by an expert surgical team.

Keywords: Esophageal perforation, esophagectomy, iatrogenic, instrumental, Ivor–Lewis


How to cite this article:
Kassab AK, Taha AY, Kaznazani K. Instrumental esophageal perforation: A case series. Arch Int Surg 2017;7:30-5

How to cite this URL:
Kassab AK, Taha AY, Kaznazani K. Instrumental esophageal perforation: A case series. Arch Int Surg [serial online] 2017 [cited 2021 May 15];7:30-5. Available from: https://www.archintsurg.org/text.asp?2017/7/1/30/229178




  Introduction Top


Esophageal perforation (EP) is a rare but potentially life-threatening condition with a morbidity and mortality greater than 20%.[1],[2],[3],[4] Iatrogenic trauma accounts for nearly 75% of the cases,[3] while the remainder can be produced by penetrating trauma, ingestion of foreign bodies, ingestion of corrosive agents, and sometimes blunt trauma. Iatrogenic EP can be produced by rigid and flexible esophagoscopy, placement of nasogastric (NG) tubes, endotracheal tubes and stents, and balloon dilatation of benign or malignant esophageal strictures. Spontaneous perforation is relatively rare and reported in 15% of cases only.[3],[5],[6],[7]

The points that make EP a serious condition are the anatomical location of the esophagus in the posterior mediastinum, proximity of vital structures, difficulty of accessing the esophagus, poor or segmental blood supply, lack of strong serosal covering, escape of esophageal contents rich in bacteria to a closed compartment producing severe mediastinitis, nonspecific symptoms and signs, late presentation, and delayed therapy. Moreover, rarity of the condition does not allow individual physicians to gain enough experience in managing this condition. Most information available in the literature is derived from case reports or small case series, and it is not possible to carry out prospective large studies.[6] Schweigert et al., on the other hand, conducted a truly exceptional multinational retrospective study that included 288 patients with EP over 25 years at 11 European centers.[1],[2]

The treatment of iatrogenic EP is controversial. However, it should be individualized according to the circumstances of each case and the experience of the surgeon.[6] Treatment options may be nonoperative [7] or operative.[3],[8],[9],[10] Different procedures described for esophageal perforation include primary repair with or without reinforcement, simple drainage of the thoracic cavity, exclusion diversion operation, and esophageal resection with or without primary reconstruction. Thoracoscopic repair using minimally invasive surgery has also been described in the literature.[3] The aim of this paper is to discuss the management of three cases of instrumental EP in view of the published literature.


  Case Reports Top


Case 1

A 4-year-old girl [Figure 1]a admitted to Sulaimaniyah Teaching Hospital (STH) on May 11, 2009 because of dysphagia following a corrosive esophageal injury. She had inadvertently ingested a bleaching agent 1 year earlier. She was initially dealt with by a pediatric gastroenterologist who performed 8 esophageal dilatations under general anesthesia (GA). During the last dilatation, she developed a perforation of esophagus. We received her in a toxic state with an acute left-sided empyema [Figure 1]b. We have managed her initially conservatively by chest tube, antibiotics, and feeding jujenostomy [Figure 1]c. Three months later, she underwent an esophageal replacement by stomach placed retrosternally (gastric pull-up). The approach was via a laparotomy and left neck incision [Figure 1]d,[Figure 1]e,[Figure 1]f,[Figure 1]g,[Figure 1]h,[Figure 1]i,[Figure 1]j,[Figure 1]k. The patient did well postoperatively. Despite slight difficulty in swallowing a solid diet, she progressively gained weight. The main complication was a chronic purulent discharge at the cervical wound (chronic discharging sinus) which required several explorations till it was finally controlled. The last follow-up visit, 7 years after operation, revealed no significant complication with satisfactory swallowing and a body weight of 29 kg.
Figure 1: (a) A 4-year-old girl with dysphagia due to corrosive esophageal injury; (b) Left-sided empyema; (c) Feeding jujenostomy; (d-k) Gastric pull-up via laparotomy and left neck incision

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Case 2

A 54-year-old man [Figure 2]a was admitted to thoracic surgery department/STH on February 26, 2005 because of dysphagia. Chest radiograph [Figure 2]b and Barium swallow [Figure 2]c were normal. Rigid esophagoscopy under GA revealed no abnormality. Unfortunately, a small perforation occurred at lower esophagus. It was immediately discovered as lung tissue could be seen through the rent. The patient started to have severe chest pain after recovery from GA. Chest radiograph soon after endoscopy [Figure 2]d was normal; however, a subsequent film [Figure 2]e revealed a small-sized left apical pneumothorax and a minimal pleural effusion. The patient and his family were informed about the complication and the need for urgent intervention. After taking the informed consent, left lateral 7th space thoracotomy was performed several hours after the perforation. A small tear was found at lower thoracic esophagus; closed by interrupted full-thickness silk sutures. NG and chest tubes were placed [Figure 2]f. The postoperative course was uneventful. Oral diet started on the 5th postoperative day when gastrograffin swallow revealed no leak.
Figure 2: (a) A 54-year-old man was admitted because of dysphagia; (b) Normal chest radiograph; (c) Normal barium esophagography; (d) Chest radiograph soon after rigid esophagoscopy looked normal; (e) Subsequent chest film revealed small-sized left apical pneumothorax and minimal pleural effusion; (f) Chest radiograph after trans-thoracic repair of esophageal tear

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Case 3

A 24-year-old man was admitted to a private surgical hospital on January 5, 2011 because of long-standing dysphagia. The patient was a known case of achalasia cardia since childhood. He underwent two transabdominal esophagomyotomies at the ages of 12 and 15 years; both were performed by a general surgeon. He had temporary improvement after the first operation but not after the second [Figure 3]a. Esophageal dilatation was done 1 month earlier. Despite this, his dysphagia was not relieved, and unfortunately, the lower thoracic esophagus was perforated during the second dilatation [Figure 3]b and [Figure 3]c. The patient developed severe chest pain and shortness of breath. He deteriorated rapidly and developed cardiac arrest three times but fortunately responded to resuscitation.
Figure 3: (a) Barium swallow showing evidence of severe achalasia cardia despite 2 trans-abdominal esophagomyotomies in a 24-year-old man; (b) Dilatation balloon; (c) Endoscopic pictures showing bleeding adjacent to the dilating balloon; (d) Note the severe surgical emphysema of the neck and the placed endotracheal tube, (e) Right thoracotomy revealed extensive surgical emphysema of the entire thoracic esophagus, (f and g) Resection of injured lower esophagus with portions of stomach and diaphragm; (h and i) Patient's photo and chest radiograph after operation

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When the thoracic surgeon was called to see him, he was in severe respiratory distress having stridor, restlessness, cyanosis, and severe surgical emphysema of the neck. The pulse was weak and rapid. The general condition was too poor to allow detailed proper assessment of the injury. The patient was taken urgently to the operating room and endotracheal tube was placed [Figure 3]d. He began to improve and regained a pink color. Needle aspiration from both pleural spaces revealed no pneumothorax. However, no portable X-ray was available to confirm that. The endoscopic pictures showed bleeding adjacent to the dilating balloon but the size of the tear was not evident.

We thought of a surgical approach to address both the tear and achalasia. Laparotomy revealed severe adhesions of the stomach to the liver and spleen as well as similar adhesions between esophagus and diaphragm. It was very difficult to release these adhesions or consider a repeat esophagomyotomy. Therefore, we decided to proceed with resection. The stomach was mobilized along the right gastric artery. Right thoracotomy revealed extensive surgical emphysema of the entire thoracic esophagus [Figure 3]e. There was a 3-cm tear in the wall of the right side of the esophagus. Distally, the esophagus felt like a hard cord. The injured lower esophagus together with a portion of stomach and diaphragm were resected [Figure 3]f and [Figure 3]g followed by gastro-esophagostomy (Ivor–Lewis operation). The postoperative course was smooth and the patient went home on the 4th postoperative day [Figure 3]h and [Figure 3]i.


  Discussion Top


Iatrogenic injury refers to an injury caused unintentionally by medical treatment. While such an injury is not considered a legal mistake if it was inflicted without a negligence,[11] it remains a very sensitive issue. Medical ethics dictate that the patient should be informed about the likelihood of occurrence of EP during diagnostic or therapeutic procedures on the esophagus and the event should be declared to the patient once it occurs. Further steps should follow to fix the problem as early and properly as possible. Failure to do so invites more deterioration and a grave outcome. Iatrogenic EP has a mortality of 10%.[3] The reported risk for EP is 0.03% in diagnostic OGD, 0.5% in esophageal dilation of all types, and 1.7% in esophageal dilation for achalasia.[3] The extreme rarity of iatrogenic EP after endoscopy implies that a physician may or may not see a single case in his/her life time. Nevertheless, he/she should be able to immediately diagnose and properly treat this serious event.

It is worth noting that the 3 EPs in this article were performed by physicians of different specialties (a gastroenterologist, thoracic surgeon, and general surgeon). However, all perforations were fixed by a thoracic surgeon. This does not imply that physicians other than thoracic surgeons are not allowed to perform esophageal interventions, but they should be part of a team involving a thoracic surgeon.

In the first patient, we think it was better to choose esophageal substitution surgery from the start rather than repeat dilatations. Nonoperative management of caustic esophageal injuries often results in esophageal stricture, necessitating long-term interventions.[12] Severe stricture formation that does not respond to dilatation is best managed by esophageal replacement.[13] The most commonly used conduits for esophageal substitution are the colon [12],[13] and the stomach. In a review of patients treated by gastric pull-up, long-term outcome was good; complications included leaking anastomosis (n = 15; 36%), which uniformly resolved without intervention, and stricture formation (n = 40; 49%), which responded to a course of dilation.[13]

When perforation has developed it should be diagnosed earlier and referred to thoracic surgery immediately. Primary repair could not be chosen due to sepsis and late presentation, and therefore, a conservative treatment was appropriately used.[3] We agree with many authors [3] that a feeding jujenostomy should always be added for nutrition.

In this child, gastric pull-up was performed via laparotomy and neck incision without thoracotomy; the diseased esophagus was not removed and this point is controversial. The long-term risk of developing cancer of esophagus is higher among patients with ingested corrosive agents than in the normal population; therefore, some authors recommend esophagectomy at the time of esophageal substitution.[14]

Although accidental caustic ingestions in children are decreasing in developed countries, they remain a prevalent problem in developing world.[12],[14] Preventive strategies should include regulation and packaging of corrosive substances, organization of psychiatric services, and education of the population on corrosive ingestion.[14]

Because of the relative rarity of esophageal caustic injuries requiring esophagectomy, there are very few studies that address the complications of surgical treatment of corrosive esophageal injuries.[12] Exception to this fact, Voron et al.[15] presented a truly remarkable series of 100 patients who required revision procedures to the esophagus injured by caustic ingestion. The authors had clearly the benefit of a large referral population of their center (Saint-Louis Hospital in Paris).[12]

In the second patient, we think it was safer to use flexible OGD as it is less traumatic than rigid esophagoscope. Perforation could have been produced by using unnecessary force. No abnormality was detected (NAD) during esophagoscopy in this case. Similarly, Lawrence et al.[16] in their series of 30 iatrogenic EPs caused by upper gastrointestinal endoscopy found no abnormality of esophagus before perforation in 15 cases (50%). Luckily, the perforation was immediately discovered at the time of endoscopy by seeing a lung tissue. Primary transthoracic repair was a good option and the outcome was excellent. This option was chosen due to early diagnosis of perforation of a previously healthy esophagus and the presence of hydropneumothorax, a usual finding in patients with distal third esophageal perforations.[3]

In the third patient, the original esophagomyotomy could have been incomplete; thus, dysphagia persisted. Fortunately, EP was diagnosed early and the thoracic surgeon was instantly consulted. Urgent esophagectomy was performed with excellent recovery. Emergency esophagectomy for perforation of the esophagus following pneumatic dilatation of esophagus in achalasia is an old procedure reported for the first time in 1953.[3],[10] The rationale for esophagectomy was the following. When we have a perforation of a diseased or distally-obstructed esophagus, then we should think of a procedure that addresses both problems, i.e., repair of perforation and relief of obstruction. Repair of EP without relieving the obstruction would result in failure, i.e., leaking repair.[3] The length of the tear was unknown and the patient's general condition did not allow more accurate assessment of the injury. The side of EP (right or left) was also unknown to us as there was no proven pneumothorax. If a thoracotomy was chosen, it would have enabled us to suture a small tear but narrowing may result from suturing a big tear. Moreover, a repeat esophagomyotomy was not possible due to dense adhesions of previous surgery. Hence, we considered resection of the injured lower esophagus together with portions of stomach and diaphragm in one piece (esophagogastrectomy) and to suture the stomach to esophagus (gastroesophagostomy). This was done via laparotomy and right thoracotomy (Ivor–Lewis operation). We agree with Braga de Aquino et al. from Brazil [9] and others [3] who conclude that, despite its morbidity, emergency esophagectomy has its validity, especially in well-indicated cases of EP subsequent to endoscopic dilation for benign strictures. Esophageal resection should be considered for perforations in patients with megaesophagus, carcinoma, caustic ingestion, stenosis, or severe reflux strictures that cannot be dilated.[3] Although endoscopic forceful pneumatic dilatation has been generalized as the primary choice of treatment for esophageal achalasia, perforation occurs in approximately 2–5% of cases. The most widely used treatment for this devastating complication is primary repair of perforation combined with esophagomyotomy on the opposite side. However, this is a complicated and time-consuming procedure. To simplify the procedure, Kim from Korea, managed 2 cases of iatrogenic EP in achalasia using gastroplasty with good results.[8]

Regarding diagnosis, the first patient was received acutely ill with fever and acute empyema. We had no idea about endoscopic findings. Chest radiography and contrast esophagogram at referral time revealed a long stricture and loculated empyema. The EP might have healed at that point in time. Both second and third patients were diagnosed at endoscopy and had severe chest pain thereafter. The third patient had surgical emphysema and respiratory distress as well. Chest pain is regarded as the cardinal symptom of EP and is present in more than 70% of the patients with a full thickness perforation of the intrathoracic esophagus. Other symptoms and signs are nonspecific. Most patients are in significant distress upon physical examination. Tachycardia is common.[6]

Most literature regarding EP emphasizes the importance of time interval between perforation and initiation of surgical therapy. The reported mortality from treated EP is 10–25% when therapy is initiated within 24 hours of perforation, but it could increase to 40–60% when the treatment is delayed beyond 48 hours.[3] Both adult patients in our study were diagnosed very soon after perforation, an important factor in their final recovery. While the little girl was referred late, a conservative management approach was initially used.

Whether an injury is iatrogenic does not matter, what matters is whether an injury was caused by negligence. Iatrogenic injury is not necessarily a result of negligence and is not necessarily an indication of incompetence.[11]


  Conclusion Top


Despite the small number of patients with EP reported here, good outcome (minimal morbidity and no mortality) is attributed to early diagnosis and timely intervention by the expert surgical team. An iatrogenic injury is not legally wrong if the doctor treats the patient with reasonable care.

Acknowledgement

We wish to thank Dr. Aram K Rash for doing feeding jujenostomy and Dr. Aram Baram for operating on the recurrent discharging neck sinus in case 1.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initial will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Schweigert M, Santos Sousa H, Solymosi N, Yankulov A, Fernández MJ, Beattie R, et al. Spotlight on esophageal perforation – A multinational study using the Pittsburgh esophageal perforation severity scoring system. J Thorac Cardiovasc Surg 2016;151:1002-11.  Back to cited text no. 1
    
2.
Antonoff MB. Validation of the Pittsburgh esophageal perforation severity score: Further impetus for a prospective study. J Thorac Cardiovasc Surg 2016;151:1012-3.  Back to cited text no. 2
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3.
Kaman L, Iqbala J, Kundilb B, Kochhar R. Management of Esophageal Perforation in Adults. Gastroenterol Res 2010;3:235-44.  Back to cited text no. 3
    
4.
Ivatury RR, Moore FA, Biffl W, Leppeniemi A, Ansaloni L, Catena F, et al. Esophageal injuries: Position paper, WSES, 2013. World J Emerg Surg 2014;9:9.  Back to cited text no. 4
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Mattox KL. The Injured Esophagus (Editorial). Texas Heart Inst J 2010;683-4.  Back to cited text no. 5
    
6.
Søreide JA, Viste A. Esophageal perforation: Diagnostic work-up and clinical decision-making in the first 24 hours. Scandinavian J Trauma Resusc Emerg Med 2011;19:66.  Back to cited text no. 6
    
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Petersen JM. The Use of a Self-Expandable Plastic Stent for an Iatrogenic Esophageal Perforation. Gastroenterol Hepatol 2010;6:389-91.  Back to cited text no. 7
    
8.
Kim H. Gastroplasty for Esophageal Perforation after Endoscopic Balloon Dilatation for Achalasia: Two Cases. J Korean Med Sci 2014;29:739-42.  Back to cited text no. 8
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9.
Braga de Aquino JL, Teixeira de Camargo JG, Cecchino GN, Pereira DA, Bento CA, Leandro-Merhi VA. Evaluation of Urgent Esophagectomy in Esophageal Perforation. Arq Bras Cir Dig 2014;27:247-50.  Back to cited text no. 9
    
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Kerr WF. Emergency esophagectomy. Thorax 1968;23:204-9.  Back to cited text no. 10
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11.
Klaas PB, Berge KH, Klaas KM, Klaas JP, Larson AN. When Patients Are Harmed, But Are Not Wronged: Ethics, Law, and History. Mayo Clin Proc 2014;89:1279-86.  Back to cited text no. 11
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Ferraris VA. Treatment of caustic esophageal injury: The source of knowledge is experience (Editorial Commentary). J Thorac Cardiovasc Surg 2016;152:1386-7.  Back to cited text no. 12
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13.
Jobe BA, Hunter JG, Peters JH. Esophagus and Diaphragmatic Hernia. In: Brunicardi FC, et al., editors. Schwartz's Principles of Surgery. 8th ed. McGraw-Hill Companies; 2005.  Back to cited text no. 13
    
14.
Adedeji TO, Tobih JE, Olaosun AO, Sogebi OA. Corrosive esophageal injuries: A preventable menace. Pan Afr Med J 2013;15:11.  Back to cited text no. 14
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15.
Voron T, Anyla M, Corte H, Roland E, Munoz-Bongrand N, Sarfati E, et al. The cervicosternolaparotomy approach for the treatment of graft dysfunction after retrosternal esophageal reconstruction for caustic injuries. J Thorac Cardiovasc Surg 2016;152:1378-85.  Back to cited text no. 15
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16.
Lawrence DR, Moxon RE, Fountain SW, Ohri SK, Townsend ER. latrogenic esophageal perforations: A clinical review. Ann R Coll Surg Engl 1998;80:115-8.  Back to cited text no. 16
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