Ann Thorac Surg 2006;81:1493-1495
© 2006 The Society of Thoracic Surgeons
Case report
Bouveret Meets Boerhaave
Biren P. Modi, MD,
Christopher Owens, MD,
Stanley W. Ashley, MD,
Yolonda L. Colson, MD, PhD*
Department of Surgery, Brigham and Women’s Hospital, Harvard Medical School, Boston, Massachussetts
Accepted for publication April 18, 2005.
* Address correspondence to Dr Colson, Department of Surgery, Brigham and Women’s Hospital, Harvard Medical School, 75 Francis St, Boston, MA 02115 (Email: ycolson{at}partners.org).
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Abstract
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Rarely, biliary-enteric fistula can result in duodenal obstruction or Bouveret’s syndrome. Boerhaave’s syndrome is a distal esophageal rupture in the setting of severe emesis. This case is the first reported successful management of these clinical scenarios occurring simultaneously and highlights important features in presentation, diagnosis, and surgical treatment.
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Introduction
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Biliary-enteric fistula is a recognized complication of cholelithiasis, occasionally producing intestinal obstruction as originally described by Courvoisier [1] in 1890. The gallstone typically obstructs the intestine in the relatively narrow terminal ileum, but can impact in the duodenum and result in gastric outlet obstruction, or Bouveret’s syndrome [2].
Nearly 300 years ago, Hermann Boerhaave described esophageal rupture resulting from the sudden increase in esophageal pressure caused by emesis [3]. The patient, Baron van Wassenaer, the Grand Admiral of the Dutch fleet, died within 24 hours of perforation.
In the current report, the successful treatment of a patient with Boerhaave’s syndrome resulting from Bouveret’s syndrome is presented and discussed. This is the first report of the successful surgical management of these two rare entities occurring simultaneously in the same patient.
An 83-year-old man presented with abdominal pain radiating to the back. For several weeks he had experienced abdominal discomfort, nausea, and heartburn. Eighteen hours prior to presentation, he had persistent emesis develop. He was transferred to our institution after chest roentgenogram revealed pneumomediastinum with right chest opacification and a question of pneumobilia (Fig 1A). Physical examination revealed tachycardia with mild epigastric tenderness. Laboratory data was significant for a white blood cell count of 13,500 per microliter. Chest computerized tomography performed at our institution revealed contrast extravasation from the distal esophagus into the right hemithorax (Fig 1B). An abdominal computed tomographic scan revealed a large gallstone within the duodenum and pneumobilia (Figs 1C, 1D).
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Fig 1. Radiographic findings at presentation. (A) Chest roentgenogram demonstrating pneumomediastinum and opacification of the right chest. (B) Computed tomography demonstrating extravasation of oral contrast from the esophagus (black arrow). (C) Computed tomography demonstrating obstructing gallstone within the duodenum (black arrow). (D) Computed tomography demonstrating pneumobilia (black arrow).
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The patient was taken to the operating room after volume resuscitation where an esophagogastroduodenoscopy revealed an esophageal tear just proximal to the gastroesophageal junction. A large obstructing gallstone was encountered within the duodenum but could not be extracted endoscopically (Fig 2). The patient subsequently underwent a combined right thoracotomy and laparotomy.
Upon entering the right hemithorax, bile staining was noted. The esophageal tear was 1.5 cm in length and extended inferiorly past the right crus of the diaphragm, which was incised radially. The esophageal defect was debrided, repaired over a T-tube, and buttressed with an intercostal muscle flap. The chest was copiously irrigated with pulse lavage and was closed with multiple chest tubes in place.
Upon entering the abdomen, significant inflammation was noted in the right upper quadrant. The stone could easily be palpated within the duodenum, but could not be extracted retrograde through a gastrotomy. The gastrotomy was extended through the pylorus and the 2.9 cm egg-shaped stone was extracted. The pyloroplasty was closed in a Heineke-Mikulicz fashion, and both gastrostomy and jejunostomy tubes were placed.
An esophogram on postoperative day 7 revealed no evidence of esophageal leak. The patient was discharged on postoperative day 14 tolerating feeding tubes plus a full liquid diet. The T-tube was clamped and removed 2 weeks later after a repeat esophogram was negative, and he was tolerating a regular diet.
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Comment
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Classically, Boerhaave’s syndrome results from an abrupt increase in intraesophageal pressure disrupting the posterolateral wall and producing hydropneumothorax. In Boerhaave’s initial description, the rupture occurred into both pleural cavities, emphasizing that either cavity can be involved [3]. Boerhaave’s syndrome was definitively fatal until the first report of successful esophageal repair by Barrett [4] in 1946, and mortality remains high at 30% to 70% in the best series. Diagnosis of Boerhaave’s syndrome can be made by clinical history and radiographic findings. Our patient presented with classical findings of pneumomediastinum and hydropneumothorax after severe emesis.
Biliary-enteric fistula is a known complication of cholelithiasis with erosion of a gallstone through the friable gallbladder wall into the duodenum. Gallstone ileus accounts for 1% to 3% of intestinal obstruction cases and as many as 20% of cases in the elderly. In 1% to 5% of these cases, Bouveret’s syndrome results from a large gallstone (
3 cm) lodged in the proximal small bowel [2]. The persistent vomiting that results can lead to life-threatening dehydration and electrolyte disturbances. Classically, Bouveret’s syndrome presents as upper abdominal pain, fever, and emesis in elderly women with a history of biliary pain. Approximately 300 cases have been reported since Leon Bouveret described the unsuccessful surgical management of 2 patients with gastric outlet obstruction in 1896. Roentgenographic diagnosis is made with Rigler’s triad, ie, gastric dilatation or small bowel dilatation with pneumobilia and an intraintestinal gallstone on computed tomography [5].
Diagnostic issues particular to this case include prompt clinical and radiologic assessment followed by endoscopic confirmation of the source of obstruction. Failure to remove the obstruction results in the breakdown of the esophageal repair. Therefore, management of both entities is paramount and includes volume resuscitation, control of soilage, treatment of esophageal rupture, extraction of the culprit stone, and placement of nutritional access.
Surgical treatment of esophageal rupture mandates control of mediastinal soilage and debridement of devitalized tissue. Closure over a T-tube allows for formation of a controlled fistula if wound healing is impaired. The hallmark of treatment is irrigation and drainage of the hemithorax to remove soilage and prevent residual collections.
Endoscopy is an important adjunct to diagnosis and is potentially therapeutic. Minimal insufflation of air, particularly in the case of gastric outlet obstruction, is utilized to prevent extension of the esophageal tear. However, failure to extract the impacted stone by endoscopy necessitates surgical intervention. Considerable controversy exists regarding the repair of the cholecystoduodenal fistula and cholecystectomy. In this elderly man, with an acute esophageal rupture and no residual gallstones, we elected not to repair the fistula. He has been without symptoms or recurrence.
We believe this is the only reported case of successful management of Boerhaave’s syndrome resulting from Bouveret’s syndrome. The only other reported case involving these two simultaneous entities was of an elderly man with epigastric pain and persistent emesis that progressed to esophageal rupture while in the emergency room. He underwent immediate surgical repair but died 1 week later [5].
In this case, a cholecystoduodenal fistula resulting in duodenal impaction of a gallstone and gastric outlet obstruction led to persistent emesis and esophageal rupture. Although these entities are rare, clinicians must have a high index of suspicion, as prompt evaluation and immediate surgical intervention are required because failure to address both entities at the initial operative setting will result in a fatal outcome.
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References
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- Courvoisier LT. Zasurstitsch-Statistiche Beitrage zur Pathologie und Chirurgie der Gallenwege. Leipzig FCW Vogel 1890.
- Cooper SG, Sherman SB, Steinhardt JE, Wilson JM, Richman AH. Bouveret’s syndrome. diagnostic considerations. JAMA 1987;258:226-228.[Abstract/Free Full Text]
- Boerhaave H. Atrocis, nec descripti prius, morbi historia. Medici, 1724. (Translated in Bull Med Libr Assoc 1955;43:217–40.).
- Barrett NR. Spontaneous perforation of the esophagus. review of the literature and report of 3 new cases. Thorax 1946;1:48.[Free Full Text]
- Rene M, Valls C, Hidalgo F, Prieto L. Duodenal gallstone ileus producing Boerhaave’s syndrome Abdom Imaging 1995;20:516-517.[Medline]
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Abstract
Introduction
