Clin J Gastroenterol (2013) 6:33–37 DOI 10.1007/s12328-012-0356-8
CASE REPORT
Recurrent spontaneous esophageal rupture Keisuke Ieta • Akira Oki • Katsunobu Teshigahara • Katsuya Osone • Shigeru Sasaki • Junichi Nakamura Koji Nakagawa • Hiroyuki Kuwano
•
Received: 8 November 2012 / Accepted: 11 December 2012 / Published online: 3 January 2013 Ó Springer Japan 2013
Abstract Spontaneous rupture of the esophagus is a relatively uncommon event, and recurrent rupture is extremely rare. We present a patient who experienced and survived 2 spontaneous perforations of the esophagus, occurring 6 years apart. A 43-year-old man was admitted to our hospital with upper abdominal pain after vomiting. Esophagoscopy, esophagogram, and computed tomography were suggestive of esophageal rupture. Emergency left thoracolaparotomy revealed a 20-mm perforation of the left lower esophageal wall that had been previously repaired. After the perforation was repaired with a single-layer closure, the mediastinum and pleural cavity were drained. The patient recovered well and was discharged from the hospital on postoperative day 29. To the best of our knowledge, only 7 previous cases of recurrent spontaneous esophageal perforation have been reported in the literature. Keywords Recurrence Spontaneous rupture Esophagus Review
Introduction Spontaneous rupture of the esophagus, also known as Boerhaave’s syndrome, is a relatively uncommon but
K. Ieta (&) A. Oki K. Teshigahara K. Osone S. Sasaki J. Nakamura K. Nakagawa Department of Surgery, Saitama Red Cross Hospital, 8-3-33 Kamiochiai, Chuo-ku, Saitama 338-8553, Japan e-mail: [email protected] H. Kuwano Department of General Surgical Science, Graduate School of Medicine, Gunma University, 3-39-22 Showa-machi, Maebashi, Gunma 371-8551, Japan
life-threatening condition; early diagnosis and prompt treatment are essential if the patient is to survive. The symptoms, however, are non-specific, making diagnosis difficult. Treatment is frequently delayed, resulting in a high mortality rate. Recurrent esophageal rupture is rarely reported. We present an unusual case of Boerhaave’s syndrome in which a patient experienced recurrent spontaneous esophageal rupture; we also review previously reported instances of this rare phenomenon.
Case report A 43-year-old man was admitted to our hospital 5 h following onset of slightly bloody vomitus after drinking alcohol. The chief complaint was gradually worsening upper abdominal pain at the time of admission. His past medical history was significant for a spontaneous esophageal rupture that had occurred 6 years earlier, which had been treated surgically via a left thoracolaparotomy. Perforation of the left lower esophagus had been detected, treated with primary closure, and covered by the greater omentum. He also had a history of pyothorax 2 years after the esophageal rupture, which had been treated on 3 separate occasions with surgical repair (thoracoscopic surgery, right thoracotomy, and thoracoplasty). Although the cause of pyothorax was unknown, there was no relation to the primary surgery of esophageal rupture. The patient’s social history was significant for moderate-to-heavy alcohol use. On admission, the patient complained of severe epigastric pain and had a heart rate of 89 beats/min, blood pressure of 109/63 mmHg, respiratory rate of 24 breaths/ min, body temperature of 36.9 °C, and oxygen saturation of 100 % on room air. He was a lean man with a height of
123
34
Clin J Gastroenterol (2013) 6:33–37
Fig. 1 a Upper endoscopy disclosing a deep laceration with clot in the left lower esophageal wall. b The laceration is closed with endoscopic clips
1.71 m and a weight of 51 kg. Physical examination revealed midepigastric tenderness without voluntary guarding; there was no subcutaneous emphysema. Blood biochemistry revealed an abnormal white blood cell count of 11,540/lL and hemoglobin of 10.7 g/dL; C-reactive protein was normal at 0.0 mg/dL. Radiographic findings included a small, left-sided pleural effusion, no pneumothorax, and no intraabdominal free air; post-thoracoplasty changes were visible, including a hyperlucent right lung and vestiges of the right seventh, eighth, ninth and tenth rib excisions. Upper gastrointestinal hemorrhage was suspected at first, and endoscopy was performed. Esophagoscopy revealed the presence of a deep laceration with a clot in the left lower esophageal wall (Fig. 1a). Effusive bleeding was detected when the clotted blood was removed with endoscopic forceps; clips were used to arrest the hemorrhage and to close the laceration to the extent possible (Fig. 1b). It was not possible to confirm if all the layers of the esophagus were damaged. A water-soluble contrast swallow revealed no obvious leakage (Fig. 2). Computed tomography revealed a left-sided pleural effusion as well as emphysema in the mediastinum around the lower esophagus (Fig. 3). These results confirmed esophageal rupture, and an emergency left thoracotomy was performed 17 h after the onset of symptoms. A thoracolaparotomy incision was made in the left sixth interspace, the same location as the prior operative incision. The left lung was found to be densely adhered to the chest wall and diaphragm. After these adhesions were taken down, it became evident that hematoma, purulent material, and gastric fluid had collected in the left thoracic cavity and mediastinum. The lower esophagus was covered with the greater omentum which had been placed there during the prior esophageal operation in order to reinforce the repair. A 20-mm perforation was identified in the left lateral wall of the distal esophagus, at the site of the previous repair (Fig. 4).
123
Fig. 2 Gastrografin esophagogram showing no leakage of contrast medium. The endoscopic clips are visible
Fig. 3 Computed tomography showing left pleural effusion and pneumomediastinum
Clin J Gastroenterol (2013) 6:33–37
35
Fig. 4 Operative findings revealing a perforation in the left lateral wall of the distal esophagus
Mucosa of the perforation site was completely closed by the endoscopic clips, and the muscle layer was then repaired with monofilament absorbable sutures. Two intramediastinal drains and a chest drain were inserted, and an open gastrostomy tube was placed. The patient was transferred to the intensive care unit, where he was extubated on postoperative day 3. His postoperative course was unremarkable, with the exception of alcohol-withdrawal delirium tremens. The patient was discharged 29 days after surgery. He experienced a complete recovery and had no dysphagia, regurgitation, or heartburn at his 6-month follow-up visit.
Discussion Spontaneous esophageal perforation was first described by the Dutch physician Hermann Boerhaave in 1724 [1]. The most commonly proposed cause is a rapid increase in intraesophageal pressure that may be precipitated by forceful vomiting, causing discoordination of esophageal motility. We were only able to locate 7 prior reports of recurrent spontaneous esophageal rupture; our patient makes a total of 8 case reports. Table 1 reviews the previously published reports on recurrent spontaneous esophageal rupture [2–7]. In 90 % of patients, esophageal rupture is localized to the left posterolateral part of the lower third of the esophagus [8]. The relative consistency of this perforation site has been attributed to an inherent anatomic weakness created by the lack of supporting structures, the penetration of nerves and vessels into the esophageal muscle wall in this area, and the angulation of the esophagus itself [9]. Although the primary rupture site in patients with recurrence is frequently the left lower thoracic esophagus (62.5 %), the recurrence site has a tendency to be on the right side (50 %) rather than the left side (25 %) of the distal esophagus. In 5 of the 8 known cases (62.5 %), the recurrent site was different from the primary site; the same site ruptured in the other 2 patients (25 %). It is likely that
recurrence occurs in the right lower esophagus, rather than the site of primary perforation, because the original rupture site has been strengthened by surgical reinforcement, adhesions, and scar formation. In our patient, recurrence presumably occurred at the same site as the primary rupture because the adhesions and scarring in the right thoracic cavity from 3 subsequent surgical treatments for right pyothorax strengthened this side over the left side. Our patient did not experience tension pneumothorax or subcutaneous emphysema, although endoscopy was performed immediately for the suspicion of upper gastrointestinal hemorrhage. We hypothesize that the aforementioned adhesions and scar tissue prevented air from spreading much beyond the perforation site. In addition, it was thought that heavy alcohol use was deeply involved as the cause of this recurrence. There were cases of alcohol and drug addiction in the previous reports of recurrent spontaneous esophageal rupture [4, 5]. The treatment of alcohol dependence and mental care are considered to be important in order to prevent recurrence. There are conservative treatments as well as surgical treatments for esophageal rupture. Cameron et al. [10] described the following criteria for conservative treatment—the esophageal rupture is confined to the mediastinum, the perforation is well-drained into the esophagus, and the patient has minimal symptoms and no evidence of clinical sepsis. Surgical treatment is usually selected in patients with rupture into the pleural cavity. Many surgical management strategies for Boerhaave’s syndrome have been described, including primary closure, lavage and drainage, reinforcement of the repaired rupture with omentum, diaphragm, pleura, or a fundic patch, controlled fistula using a T-tube [11], esophagectomy, esophagostomy, and 2-term reconstruction of the digestive tract, among others. The ‘golden period’ for closure of esophageal perforation is known to be within the first 12 h, because the likelihood of a postoperative leak increases after 24 h [12]. Of the 8 known patients with recurrence, 3 chose conservative therapy and 5 received surgical
123
Survival Surgical 20
Survival Conservative 30
Different site Same site
Survival Surgical 20
Survival Conservative 5
Survival Surgical
Vomiting, chest pain
Hematemesis, right chest pain
Right chest pain
Vomiting, hematemesis, upper abdominal pain
24 days
26 months
27 months
6 years Surgical Unknown 43
Male
Lower left
Surgical 30 Lower left Male 49
Surgical 30 Lower right Male 59
74
Male
Middle left
25
Conservative
Right chest pain 8 months Surgical 10 Lower left Male 45
30 years Surgical Unknown
8
7
6
5
4
Reeder [4] Lujan [5] Nakata [6] Khan [7] Khan [7] Our patient 3
66
Male
Lower left
Conservative Surgical Unknown 30 Saha [2] Kish [3] 1 2
17 51
Female Male
Middle Lower left
Treatment Size (mm) Location (thoracic esophagus)
20
Surgical 40
Survival Death Conservative Surgical Unknown 60
Unknown Different site Different site Different site Different site Same site
Middle Lower right Lower left Lower right Middle right Lower right Lower right Lower left Chest pain, dysphagia Right upper abdominal pain, respiratory distress Hematemesis, epigastric pain 6 months 14 months
Treatment Size (mm) Location (thoracic esophagus) Chief complaint
Recurrent rupture
Period from primary rupture Primary rupture Gender Age (years) Authors
123
treatment. The time period to recurrence in the 2 patients with original conservative treatment was within 6 months, while recurrences were delayed by a few years in patients who had surgery for the first rupture. Our patient had evidence of a free leak into the thorax. We therefore performed a thoracotomy in order to evacuate the pleural cavity and to repair the rupture. We chose to treat our patient with drainage and closure of the esophageal rupture because less than 24 h had elapsed since the onset of symptoms; in addition, the esophageal mucosal surface had been closed with endoscopic clips. Spontaneous esophageal rupture is one of the most urgent examples of gastrointestinal tract perforation; however, the diagnosis is difficult because patients typically present with a wide range of symptoms. The condition is easy to misdiagnosis [13], resulting in a high mortality rate of 20–40 % [8, 14]. Mortality rates reportedly vary from 0 % if operative treatment is started within 24 h of onset to 29 % if treatment is started later than 48 h after onset [8]. It is probably more difficult to diagnose recurrent spontaneous esophageal rupture because of the condition’s rarity, being low on the differential diagnosis list. In the reported recurrent cases, 1 patient died of complications related to surgery, but 7 of 8 patients survived. A good prognosis can seemingly be obtained with early diagnosis and treatment. In summary, we present a rare case of recurrent spontaneous esophageal rupture. Successful management of recurrent rupture depends on early diagnosis and prompt treatment. Conflict of interest The authors declare that they have no conflict of interest.
Case
Table 1 Review of reported recurrent spontaneous esophageal ruptures
Survival
Clin J Gastroenterol (2013) 6:33–37 Outcome
36
References 1. Derbes VJ, Mitchell RE Jr. Hermann Boerhaave’s Atrocis, nec descripti prius, morbi historia, the first translation of the classic case report of rupture of the esophagus, with annotations. Bull Med Libr Assoc. 1955;43:217–40. 2. Saha SP, Mayo P, Long GA, Combs BF. Recurrent spontaneous perforation of the midesophagus. South Med J. 1979;72:1028–9. 3. Kish GF, Katske FA. A case of recurrent Boerhaave’s syndrome. W V Med J. 1980;76:27–30. 4. Reeder LB, Warren SE, Ferguson MK. Recurrent spontaneous perforation of the esophagus. Ann Thorac Surg. 1995;59:221–2. 5. Lujan HJ, Lin PH, Boghossian SP, Yario RF, Tatooles CJ. Recurrent spontaneous rupture of the esophagus: an unusual late complication of Boerhaave’s syndrome. Surgery. 1997;122:634–6. 6. Nakata Y, Ishii T, Tomioka N. A case of spontaneous esophageal rupture which relapsed after conservative therapy (in Japanese with English abstract). Jpn J Gastroenterol Surg. 2001;34:91–4. 7. Khan OA, Barlow CW, Weeden DF, Amer KM. Recurrent spontaneous esophageal rupture. Eur J Cardiothorac Surg. 2005;28:178–9. 8. de Schipper JP, Pull ter Gunne AF, Oostvogel HJ, van Laarhoven CJ. Spontaneous rupture of the oesophagus: Boerhaave’s
Clin J Gastroenterol (2013) 6:33–37 syndrome in 2008. Literature review and treatment algorithm. Dig Surg. 2008;2009(26):1–6. 9. Bennett DJ, Deveridge RJ, Wright JS. Spontaneous rupture of the esophagus: a review with reports of six cases. Surgery. 1970;68:766–70. 10. Cameron JL, Kieffer RF, Hendrix TR, Mehigan DG, Baker RR. Selective nonoperative management of contained intrathoracic esophageal disruptions. Ann Thorac Surg. 1979;27:404–8. 11. Ojima H, Kuwano H, Sasaki S, Fujisawa T, Ishibashi Y. Successful late management of spontaneous esophageal rupture using
37 T-tube mediastinoabdominal drainage. Am J Surg. 2001;182:192–6. 12. Cho S, Jheon S, Ryu KM, Lee EB. Primary esophageal repair in Boerhaave’s syndrome. Dis Esophagus. 2008;21:660–3. 13. Lemke T, Jagminas L. Spontaneous esophageal rupture: a frequently missed diagnosis. Am Surg. 1999;65:449–52. 14. Ochiai T, Hiranuma S, Takiguchi N, Ito K, Maruyama M, Nagahama T, et al. Treatment strategy for Boerhaave’s syndrome. Dis Esophagus. 2004;17:98–103.
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CASE REPORT
Recurrent spontaneous esophageal rupture Keisuke Ieta • Akira Oki • Katsunobu Teshigahara • Katsuya Osone • Shigeru Sasaki • Junichi Nakamura Koji Nakagawa • Hiroyuki Kuwano
•
Received: 8 November 2012 / Accepted: 11 December 2012 / Published online: 3 January 2013 Ó Springer Japan 2013
Abstract Spontaneous rupture of the esophagus is a relatively uncommon event, and recurrent rupture is extremely rare. We present a patient who experienced and survived 2 spontaneous perforations of the esophagus, occurring 6 years apart. A 43-year-old man was admitted to our hospital with upper abdominal pain after vomiting. Esophagoscopy, esophagogram, and computed tomography were suggestive of esophageal rupture. Emergency left thoracolaparotomy revealed a 20-mm perforation of the left lower esophageal wall that had been previously repaired. After the perforation was repaired with a single-layer closure, the mediastinum and pleural cavity were drained. The patient recovered well and was discharged from the hospital on postoperative day 29. To the best of our knowledge, only 7 previous cases of recurrent spontaneous esophageal perforation have been reported in the literature. Keywords Recurrence Spontaneous rupture Esophagus Review
Introduction Spontaneous rupture of the esophagus, also known as Boerhaave’s syndrome, is a relatively uncommon but
K. Ieta (&) A. Oki K. Teshigahara K. Osone S. Sasaki J. Nakamura K. Nakagawa Department of Surgery, Saitama Red Cross Hospital, 8-3-33 Kamiochiai, Chuo-ku, Saitama 338-8553, Japan e-mail: [email protected] H. Kuwano Department of General Surgical Science, Graduate School of Medicine, Gunma University, 3-39-22 Showa-machi, Maebashi, Gunma 371-8551, Japan
life-threatening condition; early diagnosis and prompt treatment are essential if the patient is to survive. The symptoms, however, are non-specific, making diagnosis difficult. Treatment is frequently delayed, resulting in a high mortality rate. Recurrent esophageal rupture is rarely reported. We present an unusual case of Boerhaave’s syndrome in which a patient experienced recurrent spontaneous esophageal rupture; we also review previously reported instances of this rare phenomenon.
Case report A 43-year-old man was admitted to our hospital 5 h following onset of slightly bloody vomitus after drinking alcohol. The chief complaint was gradually worsening upper abdominal pain at the time of admission. His past medical history was significant for a spontaneous esophageal rupture that had occurred 6 years earlier, which had been treated surgically via a left thoracolaparotomy. Perforation of the left lower esophagus had been detected, treated with primary closure, and covered by the greater omentum. He also had a history of pyothorax 2 years after the esophageal rupture, which had been treated on 3 separate occasions with surgical repair (thoracoscopic surgery, right thoracotomy, and thoracoplasty). Although the cause of pyothorax was unknown, there was no relation to the primary surgery of esophageal rupture. The patient’s social history was significant for moderate-to-heavy alcohol use. On admission, the patient complained of severe epigastric pain and had a heart rate of 89 beats/min, blood pressure of 109/63 mmHg, respiratory rate of 24 breaths/ min, body temperature of 36.9 °C, and oxygen saturation of 100 % on room air. He was a lean man with a height of
123
34
Clin J Gastroenterol (2013) 6:33–37
Fig. 1 a Upper endoscopy disclosing a deep laceration with clot in the left lower esophageal wall. b The laceration is closed with endoscopic clips
1.71 m and a weight of 51 kg. Physical examination revealed midepigastric tenderness without voluntary guarding; there was no subcutaneous emphysema. Blood biochemistry revealed an abnormal white blood cell count of 11,540/lL and hemoglobin of 10.7 g/dL; C-reactive protein was normal at 0.0 mg/dL. Radiographic findings included a small, left-sided pleural effusion, no pneumothorax, and no intraabdominal free air; post-thoracoplasty changes were visible, including a hyperlucent right lung and vestiges of the right seventh, eighth, ninth and tenth rib excisions. Upper gastrointestinal hemorrhage was suspected at first, and endoscopy was performed. Esophagoscopy revealed the presence of a deep laceration with a clot in the left lower esophageal wall (Fig. 1a). Effusive bleeding was detected when the clotted blood was removed with endoscopic forceps; clips were used to arrest the hemorrhage and to close the laceration to the extent possible (Fig. 1b). It was not possible to confirm if all the layers of the esophagus were damaged. A water-soluble contrast swallow revealed no obvious leakage (Fig. 2). Computed tomography revealed a left-sided pleural effusion as well as emphysema in the mediastinum around the lower esophagus (Fig. 3). These results confirmed esophageal rupture, and an emergency left thoracotomy was performed 17 h after the onset of symptoms. A thoracolaparotomy incision was made in the left sixth interspace, the same location as the prior operative incision. The left lung was found to be densely adhered to the chest wall and diaphragm. After these adhesions were taken down, it became evident that hematoma, purulent material, and gastric fluid had collected in the left thoracic cavity and mediastinum. The lower esophagus was covered with the greater omentum which had been placed there during the prior esophageal operation in order to reinforce the repair. A 20-mm perforation was identified in the left lateral wall of the distal esophagus, at the site of the previous repair (Fig. 4).
123
Fig. 2 Gastrografin esophagogram showing no leakage of contrast medium. The endoscopic clips are visible
Fig. 3 Computed tomography showing left pleural effusion and pneumomediastinum
Clin J Gastroenterol (2013) 6:33–37
35
Fig. 4 Operative findings revealing a perforation in the left lateral wall of the distal esophagus
Mucosa of the perforation site was completely closed by the endoscopic clips, and the muscle layer was then repaired with monofilament absorbable sutures. Two intramediastinal drains and a chest drain were inserted, and an open gastrostomy tube was placed. The patient was transferred to the intensive care unit, where he was extubated on postoperative day 3. His postoperative course was unremarkable, with the exception of alcohol-withdrawal delirium tremens. The patient was discharged 29 days after surgery. He experienced a complete recovery and had no dysphagia, regurgitation, or heartburn at his 6-month follow-up visit.
Discussion Spontaneous esophageal perforation was first described by the Dutch physician Hermann Boerhaave in 1724 [1]. The most commonly proposed cause is a rapid increase in intraesophageal pressure that may be precipitated by forceful vomiting, causing discoordination of esophageal motility. We were only able to locate 7 prior reports of recurrent spontaneous esophageal rupture; our patient makes a total of 8 case reports. Table 1 reviews the previously published reports on recurrent spontaneous esophageal rupture [2–7]. In 90 % of patients, esophageal rupture is localized to the left posterolateral part of the lower third of the esophagus [8]. The relative consistency of this perforation site has been attributed to an inherent anatomic weakness created by the lack of supporting structures, the penetration of nerves and vessels into the esophageal muscle wall in this area, and the angulation of the esophagus itself [9]. Although the primary rupture site in patients with recurrence is frequently the left lower thoracic esophagus (62.5 %), the recurrence site has a tendency to be on the right side (50 %) rather than the left side (25 %) of the distal esophagus. In 5 of the 8 known cases (62.5 %), the recurrent site was different from the primary site; the same site ruptured in the other 2 patients (25 %). It is likely that
recurrence occurs in the right lower esophagus, rather than the site of primary perforation, because the original rupture site has been strengthened by surgical reinforcement, adhesions, and scar formation. In our patient, recurrence presumably occurred at the same site as the primary rupture because the adhesions and scarring in the right thoracic cavity from 3 subsequent surgical treatments for right pyothorax strengthened this side over the left side. Our patient did not experience tension pneumothorax or subcutaneous emphysema, although endoscopy was performed immediately for the suspicion of upper gastrointestinal hemorrhage. We hypothesize that the aforementioned adhesions and scar tissue prevented air from spreading much beyond the perforation site. In addition, it was thought that heavy alcohol use was deeply involved as the cause of this recurrence. There were cases of alcohol and drug addiction in the previous reports of recurrent spontaneous esophageal rupture [4, 5]. The treatment of alcohol dependence and mental care are considered to be important in order to prevent recurrence. There are conservative treatments as well as surgical treatments for esophageal rupture. Cameron et al. [10] described the following criteria for conservative treatment—the esophageal rupture is confined to the mediastinum, the perforation is well-drained into the esophagus, and the patient has minimal symptoms and no evidence of clinical sepsis. Surgical treatment is usually selected in patients with rupture into the pleural cavity. Many surgical management strategies for Boerhaave’s syndrome have been described, including primary closure, lavage and drainage, reinforcement of the repaired rupture with omentum, diaphragm, pleura, or a fundic patch, controlled fistula using a T-tube [11], esophagectomy, esophagostomy, and 2-term reconstruction of the digestive tract, among others. The ‘golden period’ for closure of esophageal perforation is known to be within the first 12 h, because the likelihood of a postoperative leak increases after 24 h [12]. Of the 8 known patients with recurrence, 3 chose conservative therapy and 5 received surgical
123
Survival Surgical 20
Survival Conservative 30
Different site Same site
Survival Surgical 20
Survival Conservative 5
Survival Surgical
Vomiting, chest pain
Hematemesis, right chest pain
Right chest pain
Vomiting, hematemesis, upper abdominal pain
24 days
26 months
27 months
6 years Surgical Unknown 43
Male
Lower left
Surgical 30 Lower left Male 49
Surgical 30 Lower right Male 59
74
Male
Middle left
25
Conservative
Right chest pain 8 months Surgical 10 Lower left Male 45
30 years Surgical Unknown
8
7
6
5
4
Reeder [4] Lujan [5] Nakata [6] Khan [7] Khan [7] Our patient 3
66
Male
Lower left
Conservative Surgical Unknown 30 Saha [2] Kish [3] 1 2
17 51
Female Male
Middle Lower left
Treatment Size (mm) Location (thoracic esophagus)
20
Surgical 40
Survival Death Conservative Surgical Unknown 60
Unknown Different site Different site Different site Different site Same site
Middle Lower right Lower left Lower right Middle right Lower right Lower right Lower left Chest pain, dysphagia Right upper abdominal pain, respiratory distress Hematemesis, epigastric pain 6 months 14 months
Treatment Size (mm) Location (thoracic esophagus) Chief complaint
Recurrent rupture
Period from primary rupture Primary rupture Gender Age (years) Authors
123
treatment. The time period to recurrence in the 2 patients with original conservative treatment was within 6 months, while recurrences were delayed by a few years in patients who had surgery for the first rupture. Our patient had evidence of a free leak into the thorax. We therefore performed a thoracotomy in order to evacuate the pleural cavity and to repair the rupture. We chose to treat our patient with drainage and closure of the esophageal rupture because less than 24 h had elapsed since the onset of symptoms; in addition, the esophageal mucosal surface had been closed with endoscopic clips. Spontaneous esophageal rupture is one of the most urgent examples of gastrointestinal tract perforation; however, the diagnosis is difficult because patients typically present with a wide range of symptoms. The condition is easy to misdiagnosis [13], resulting in a high mortality rate of 20–40 % [8, 14]. Mortality rates reportedly vary from 0 % if operative treatment is started within 24 h of onset to 29 % if treatment is started later than 48 h after onset [8]. It is probably more difficult to diagnose recurrent spontaneous esophageal rupture because of the condition’s rarity, being low on the differential diagnosis list. In the reported recurrent cases, 1 patient died of complications related to surgery, but 7 of 8 patients survived. A good prognosis can seemingly be obtained with early diagnosis and treatment. In summary, we present a rare case of recurrent spontaneous esophageal rupture. Successful management of recurrent rupture depends on early diagnosis and prompt treatment. Conflict of interest The authors declare that they have no conflict of interest.
Case
Table 1 Review of reported recurrent spontaneous esophageal ruptures
Survival
Clin J Gastroenterol (2013) 6:33–37 Outcome
36
References 1. Derbes VJ, Mitchell RE Jr. Hermann Boerhaave’s Atrocis, nec descripti prius, morbi historia, the first translation of the classic case report of rupture of the esophagus, with annotations. Bull Med Libr Assoc. 1955;43:217–40. 2. Saha SP, Mayo P, Long GA, Combs BF. Recurrent spontaneous perforation of the midesophagus. South Med J. 1979;72:1028–9. 3. Kish GF, Katske FA. A case of recurrent Boerhaave’s syndrome. W V Med J. 1980;76:27–30. 4. Reeder LB, Warren SE, Ferguson MK. Recurrent spontaneous perforation of the esophagus. Ann Thorac Surg. 1995;59:221–2. 5. Lujan HJ, Lin PH, Boghossian SP, Yario RF, Tatooles CJ. Recurrent spontaneous rupture of the esophagus: an unusual late complication of Boerhaave’s syndrome. Surgery. 1997;122:634–6. 6. Nakata Y, Ishii T, Tomioka N. A case of spontaneous esophageal rupture which relapsed after conservative therapy (in Japanese with English abstract). Jpn J Gastroenterol Surg. 2001;34:91–4. 7. Khan OA, Barlow CW, Weeden DF, Amer KM. Recurrent spontaneous esophageal rupture. Eur J Cardiothorac Surg. 2005;28:178–9. 8. de Schipper JP, Pull ter Gunne AF, Oostvogel HJ, van Laarhoven CJ. Spontaneous rupture of the oesophagus: Boerhaave’s
Clin J Gastroenterol (2013) 6:33–37 syndrome in 2008. Literature review and treatment algorithm. Dig Surg. 2008;2009(26):1–6. 9. Bennett DJ, Deveridge RJ, Wright JS. Spontaneous rupture of the esophagus: a review with reports of six cases. Surgery. 1970;68:766–70. 10. Cameron JL, Kieffer RF, Hendrix TR, Mehigan DG, Baker RR. Selective nonoperative management of contained intrathoracic esophageal disruptions. Ann Thorac Surg. 1979;27:404–8. 11. Ojima H, Kuwano H, Sasaki S, Fujisawa T, Ishibashi Y. Successful late management of spontaneous esophageal rupture using
37 T-tube mediastinoabdominal drainage. Am J Surg. 2001;182:192–6. 12. Cho S, Jheon S, Ryu KM, Lee EB. Primary esophageal repair in Boerhaave’s syndrome. Dis Esophagus. 2008;21:660–3. 13. Lemke T, Jagminas L. Spontaneous esophageal rupture: a frequently missed diagnosis. Am Surg. 1999;65:449–52. 14. Ochiai T, Hiranuma S, Takiguchi N, Ito K, Maruyama M, Nagahama T, et al. Treatment strategy for Boerhaave’s syndrome. Dis Esophagus. 2004;17:98–103.
123
![[Spontaneous esophageal rupture].](/assets/img/hold.png)
