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Ann Thorac Surg                                                                             CASE REPORT  ASHRAFI ET AL             317
2007;83:317–9                                                            MINIMALLY INVASIVE MANAGEMENT OF BOERHAAVE’S




occur ectopically, affecting the neck, middle or poste-                  sented to the emergency department with vomiting,
rior mediastinum, and lung [5, 6]. However, ectopic                      followed by severe retrosternal and epigastric pain of
thymoma occurring in the pleura is extremely rare and                    sudden onset. An esophagogram showed evidence of
has been infrequently documented [7].                                    free extravasation of contrast from the left posterolateral
  The differential diagnoses for giant intrathoracic                     aspect of the distal esophagus just above the level of the
mass are a pleural tumor (e.g., solitary fibrous tumor,                   hiatus. A minimally invasive technique was used to
malignant mesothelioma, and sarcomas), a chest wall                      repair this injury.
tumor, or a metastatic mass. MRI findings of the                                                   (Ann Thorac Surg 2007;83:317–9)
thymoma have the same or slightly increased intensity                                 © 2007 by The Society of Thoracic Surgeons
as that of muscle on T1-weighted images and increased
intensity on T2-weighted images. Inhomogeneous sig-
nal intensity on T2-weighted images with a lobulated
border, fibrous band, and lobulated internal architec-
                                                                         B   oerhaave’s syndrome is associated with a significant
                                                                              risk of mortality and morbidity. Prompt surgical
                                                                         management is the treatment of choice. The accepted
ture is indicative of an invasive thymoma [8]. Although                  management involves surgical repair of the perforation
the MRI features of this case resembled those of                         using a thoracotomy or laparotomy, or both. Reducing
orthotopic thymoma, preoperative diagnosis was diffi-                     the inflammatory response by minimizing the surgical
cult because of the unusual location.                                    trauma may decrease the mortality risk of this potentially
  In summary, this report documents an extremely rare                    lethal condition. We report the successful laparoscopic
occurrence of ectopic pleural thymoma presenting as a                    and thoracoscopic management of a patient with Boer-
giant mass in the thoracic cavity.                                       haave’s syndrome. Although open repair and drainage




                                                                                                                                          FEATURE ARTICLES
                                                                         are the gold standard, we conclude that laparoscopic and
References                                                               thoracoscopic management of Boerhaave’s syndrome is a
                                                                         feasible alternative.
1. Rosai J, Sobin LH. Histological typing of tumors of the
   thymus, 2nd ed. New York: Springer, 1999.
2. Richardson MA, Sie KYC. The neck: embryology and anat-                A 42-year-old man with a long-standing history of inter-
   omy, 3rd ed. Philadelphia: WB Saunders Co, 1996.                      mittent dysphagia that required a change in the patient’s
3. Rosai J, Levine GD. Tumors of the thymus, 2nd ed. Washing-            dietary habits presented to the emergency department
   ton: Armed Forces Institute Pathol, 1976.
4. Detterbeck FC, Parsons AM. Thymic tumors. Ann Thorac                  with a 5-hour history of vomiting, followed by severe
   Surg 2004;77:1860 –9.                                                 retrosternal and epigastric pain of sudden onset. On
5. Moran CA, Suster S, Fishback NF, Koss MN. Primary in-                 initial presentation, his blood pressure was 142/86, pulse
   trapulmonary thymoma: a clinicopathologic and immunohis-              was 100/min, and his respiratory rate was 22/min. The
   tochemical study of eight cases. Am J Surg Pathol 1995;19:
   304 –12.                                                              patient was afebrile and mildly distressed.
6. Minniti S, Valentini M, Pinali L, Malago R, Lestani M,                   On chest exam, there was decreased air entry over the
   Procacci C. Thymic masses of the middle mediastinum: report           left hemithorax, with crackles at the left lung base. His
   of 2 cases and review of the literature. J Thorac Imag 2004;19:       abdominal exam revealed a nondistended abdomen and
   192–5.
7. Moran CA, Travis WD, Rosado-de-Christenson M, Koss MN,
                                                                         epigastric tenderness without generalized peritonitis.
   Rosai J. Thymomas presenting as pleural tumors: report of             The leucocyte count on admission was 12.1 ϫ 109/L. The
   eight cases. Am J Surg Pathol 1992;16:138 – 44.                       initial chest radiograph revealed a small left pleural
8. Kushihashi T, Fujisawa H, Munechika H. Magnetic resonance             effusion. A contrast-enhanced computed tomography
   imaging of thymic epithelial tumors. Crit Rev Diagn Imag
                                                                         (CT) scan of the chest demonstrated pneumomediasti-
   1996;37:191–259.
                                                                         num and a left pleural effusion highly suggestive of
                                                                         esophageal perforation (Fig 1). The result of a CT scan of
                                                                         the abdomen was normal. A Gastrografin (Tyco/
Minimally Invasive Management                                            MallinKrodt, St. Louis, MO) swallow demonstrated free
of Boerhaave’s Syndrome                                                  extravasation of contrast from the left posterolateral
Ahmad S. Ashrafi, MD, Omar Awais, DO,                                     aspect of the distal esophagus just above the level of the
and Miguel Alvelo-Rivera, MD                                             hiatus (Fig 2).
                                                                            After aggressive volume resuscitation, commencement
The Heart Lung and Esophageal Surgery Institute, University
of Pittsburgh Medical Center, Pittsburgh, Pennsylvania                   of broad-spectrum antibiotics, and analgesia, the patient
                                                                         was taken to the operating room. On-table endoscopy
                                                                         revealed a 2-cm to 3-cm perforation just above a nar-
We report the case of a 42-year-old man with Boerhaave’s
                                                                         rowed gastroesophageal junction. A laparoscopic explo-
syndrome. His medical history was significant only for a
                                                                         ration showed no intraabdominal pathology.
long-standing history of dysphagia. The patient pre-
                                                                            We then harvested a generous portion length of the
Accepted for publication May 24, 2006.
                                                                         greater omentum and secured it to the edges of the left
                                                                         crus. We also performed a Heller myotomy given the
Address correspondence to Dr Alvelo-Rivera, The Heart, Lung and
Esophageal Surgery Institute, University of Pittsburgh Medical Center,
                                                                         patient’s long-standing history of dysphagia. A laparo-
200 Lothrop St, C-800, Pittsburgh, PA 15213; e-mail: alveloriveram@      scopic gastrostomy and feeding jejunostomy were per-
upmc.edu.                                                                formed, and the port sites were closed.

© 2007 by The Society of Thoracic Surgeons                                                                          0003-4975/07/$32.00
Published by Elsevier Inc                                                                          doi:10.1016/j.athoracsur.2006.05.111
318     CASE REPORT   ASHRAFI ET AL                                                                                      Ann Thorac Surg
                           MINIMALLY INVASIVE MANAGEMENT OF BOERHAAVE’S                                                                        2007;83:317–9




                   Fig 1. Computed tomography image of the chest shows pneumome-
                   diastinum with air tracking laterally towards the left pleural space
                   and a left pleural effusion.
FEATURE ARTICLES




                      A double-lumen endotracheal tube was then inserted,
                   and the patient was positioned in the right lateral decu-
                   bitus position for video-assisted thoracoscopic explora-
                   tion of the left chest. The esophageal perforation site was
                   identified at a level just above the esophageal hiatus. A
                   two-layer repair was performed using simple interrupted
                   sutures. The harvested omentum was then used to cover
                   the entire length of the repaired esophagus. Intraopera-
                   tive insufflation of the esophagus did not show a leak,                 Fig 3. A postoperative barium swallow shows essentially a normal
                                                                                          esophagus.



                                                                                          and we placed two chest tubes and two Jackson-Pratt
                                                                                          drains. Incisions were closed and dressings were applied.
                                                                                            The patient was transferred to the intensive care unit
                                                                                          and had an uneventful course. He was discharged to the
                                                                                          ward the next day. A contrast study on postoperative day
                                                                                          4 showed no leak or obstruction (Fig 3). He was dis-
                                                                                          charged home on postoperative day 9.


                                                                                          Comment
                                                                                          Boerhaave’s syndrome, or spontaneous (postemetic) per-
                                                                                          foration of the esophagus, was first described by Her-
                                                                                          mann Boerhaave in 1724 [1]. It is a very uncommon
                                                                                          entity, with an estimated incidence in the literature of 1 in
                                                                                          6000 patients [2]. The esophagus differs from the rest of
                                                                                          the alimentary tract in that it lacks a serosal layer, which
                                                                                          normally contains collagen and elastic fibers. This makes
                                                                                          it more susceptible to rupture at lower pressures than the
                                                                                          rest of the gastrointestinal tract. Spontaneous esophageal
                                                                                          rupture is well documented as a postemetic phenome-
                                                                                          non. Early recognition and prompt treatment are impor-
                                                                                          tant factors in minimizing the mortality. The mortality
                                                                                          ranges from 20% to 30% [3], but if left untreated, ap-
                                                                                          proaches 100%. Barrett reported the first case of success-
                                                                                          ful surgical repair in 1947 [4].
                   Fig 2. A Gastrografin swallow shows extravasation of contrast in           The minimally invasive technique could be used only if
                   the left chest.                                                        the patient is hemodynamically stable, without signs of
Ann Thorac Surg                                                                  CASE REPORT  ASHRAFI ET AL                  319
2007;83:317–9                                                 MINIMALLY INVASIVE MANAGEMENT OF BOERHAAVE’S




escalating sepsis, without significant medical risk factors    standpoint if the presentation is not acute, the perfora-
that would preclude major surgery, and in patients with       tion is well contained with good distal flow of contrast,
no contraindications for laparoscopy or thoracoscopy.         and in a patient with no signs of sepsis.
   In managing this patient, we began with laparoscopy to        Postoperative morbidity includes the non-procedure-
harvest the omentum, performed a gastrostomy, a feed-         related postoperative complications, stricture formation,
ing jejunostomy, and an esophagomyotomy. We also              leak requiring further surgical management, or diver-
wanted to assess potential intraabdominal extent of the       sion/esophagectomy, with or without delayed
injury.                                                       reconstruction.
   The repair was begun with a myotomy to identify the           A Medline search of the literature pertaining to the
true apices of the perforation. We then débrided the          minimally invasive management of Boerhaave’s syn-
nonviable tissue and performed an interrupted mucosal         drome yielded two reports. The first describes a left
repair by using absorbable suture material and a second       thoracoscopic intracorporeal suture repair and drainage.
layer of repair by approximating the esophageal muscle.       The patient had developed a leak that was managed
We then covered the repair with omentum. Other op-            conservatively [5]. The second report describes a laparo-
tions for buttressing include pleura, intercostal muscles,    scopic primary repair and a 270° posterior fundoplication
pericardial fat pad, or latissimus/serratus/pectoralis        in a 72-year-old man. The patient was discharged home
muscle.                                                       after 2 weeks of hospitalization with no leak [6].
   Jackson-Pratt drains were used for management of              Although open repair and drainage are the gold stan-
potential postoperative leak to act as a controlled fistula,   dard, we conclude that laparoscopic and thoracoscopic
as this was a case of nonconventional surgical manage-        management of Boerhaave’s syndrome is a feasible
ment. It is possible to use alternative methods of drain-




                                                                                                                                    FEATURE ARTICLES
                                                              alternative.
age (eg, chest tube) or none at all, but we prefer to drain
locally with Jackson-Pratt drains.
   Gastric decompression and nutritional support are          References
important aspects of the postoperative management.            1. Derbes VJ, Mitchell RE Jr. Herman Boerhaave Atrocis, nec
Although a nasogastric tube is an alternative, we rou-           descripti prius, morbi historia; the first translation of classic
tinely perform laparoscopic jejunostomy and gastros-             case report of rupture of the esophagus, with annotations.
                                                                 Bull Med Libr Assoc 1955;43:217–24.
tomy tubes for other conditions, and usually it only adds
                                                              2. Lillington GA, Bernatz PE. Spontaneous perforation of esoph-
15 to 20 minutes to the operating time. This allows early        agus. Dis Chest 1961;39:177– 84.
institution of enteral feeding as well as a more secure way   3. Jougon J, Mc Bride T, Delcambre F, Minniti A, Velly JF.
of keeping the stomach decompressed. Also, in the event          Primary esophageal repair for Boerhaave’s syndrome what-
of a prolonged course, it provides a more comfortable            ever the free interval between perforation and treatment. Eur
                                                                 J Cardiothorac Surg 2004;25:475–9.
drainage technique for the patient. Every patient under-      4. Barrett NR. Report of a case of spontaneous perforation of the
going surgical management of Boerhaave’s syndrome                esophagus successfully treated by operation. Br J Surg 1947;
runs the risk of leak or delayed healing, or both. There-        35:216.
fore, the feeding tube ensures optimal enteral nutrition      5. Scott HJ, Rosin RD. Thoracoscopic repair of a transmural
in the event that the patient is not able to eat in the          rupture of the oesophagus (Boerhaave’s syndrome). J R Soc
                                                                 Med 1995;88:414P–5P.
postoperative period.                                         6. Landen S, El Nakadi I. Minimally invasive approach to
   We recommend conservative treatment to patients               Boerhaave’s syndrome: a pilot study of three cases. Surg
who would not tolerate an operation from a medical               Endosc 2002;16:1354 –7.

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Minimally invasive management of boerhaave´s syndrome

  • 1. Ann Thorac Surg CASE REPORT ASHRAFI ET AL 317 2007;83:317–9 MINIMALLY INVASIVE MANAGEMENT OF BOERHAAVE’S occur ectopically, affecting the neck, middle or poste- sented to the emergency department with vomiting, rior mediastinum, and lung [5, 6]. However, ectopic followed by severe retrosternal and epigastric pain of thymoma occurring in the pleura is extremely rare and sudden onset. An esophagogram showed evidence of has been infrequently documented [7]. free extravasation of contrast from the left posterolateral The differential diagnoses for giant intrathoracic aspect of the distal esophagus just above the level of the mass are a pleural tumor (e.g., solitary fibrous tumor, hiatus. A minimally invasive technique was used to malignant mesothelioma, and sarcomas), a chest wall repair this injury. tumor, or a metastatic mass. MRI findings of the (Ann Thorac Surg 2007;83:317–9) thymoma have the same or slightly increased intensity © 2007 by The Society of Thoracic Surgeons as that of muscle on T1-weighted images and increased intensity on T2-weighted images. Inhomogeneous sig- nal intensity on T2-weighted images with a lobulated border, fibrous band, and lobulated internal architec- B oerhaave’s syndrome is associated with a significant risk of mortality and morbidity. Prompt surgical management is the treatment of choice. The accepted ture is indicative of an invasive thymoma [8]. Although management involves surgical repair of the perforation the MRI features of this case resembled those of using a thoracotomy or laparotomy, or both. Reducing orthotopic thymoma, preoperative diagnosis was diffi- the inflammatory response by minimizing the surgical cult because of the unusual location. trauma may decrease the mortality risk of this potentially In summary, this report documents an extremely rare lethal condition. We report the successful laparoscopic occurrence of ectopic pleural thymoma presenting as a and thoracoscopic management of a patient with Boer- giant mass in the thoracic cavity. haave’s syndrome. Although open repair and drainage FEATURE ARTICLES are the gold standard, we conclude that laparoscopic and References thoracoscopic management of Boerhaave’s syndrome is a feasible alternative. 1. Rosai J, Sobin LH. Histological typing of tumors of the thymus, 2nd ed. New York: Springer, 1999. 2. Richardson MA, Sie KYC. The neck: embryology and anat- A 42-year-old man with a long-standing history of inter- omy, 3rd ed. Philadelphia: WB Saunders Co, 1996. mittent dysphagia that required a change in the patient’s 3. Rosai J, Levine GD. Tumors of the thymus, 2nd ed. Washing- dietary habits presented to the emergency department ton: Armed Forces Institute Pathol, 1976. 4. Detterbeck FC, Parsons AM. Thymic tumors. Ann Thorac with a 5-hour history of vomiting, followed by severe Surg 2004;77:1860 –9. retrosternal and epigastric pain of sudden onset. On 5. Moran CA, Suster S, Fishback NF, Koss MN. Primary in- initial presentation, his blood pressure was 142/86, pulse trapulmonary thymoma: a clinicopathologic and immunohis- was 100/min, and his respiratory rate was 22/min. The tochemical study of eight cases. Am J Surg Pathol 1995;19: 304 –12. patient was afebrile and mildly distressed. 6. Minniti S, Valentini M, Pinali L, Malago R, Lestani M, On chest exam, there was decreased air entry over the Procacci C. Thymic masses of the middle mediastinum: report left hemithorax, with crackles at the left lung base. His of 2 cases and review of the literature. J Thorac Imag 2004;19: abdominal exam revealed a nondistended abdomen and 192–5. 7. Moran CA, Travis WD, Rosado-de-Christenson M, Koss MN, epigastric tenderness without generalized peritonitis. Rosai J. Thymomas presenting as pleural tumors: report of The leucocyte count on admission was 12.1 ϫ 109/L. The eight cases. Am J Surg Pathol 1992;16:138 – 44. initial chest radiograph revealed a small left pleural 8. Kushihashi T, Fujisawa H, Munechika H. Magnetic resonance effusion. A contrast-enhanced computed tomography imaging of thymic epithelial tumors. Crit Rev Diagn Imag (CT) scan of the chest demonstrated pneumomediasti- 1996;37:191–259. num and a left pleural effusion highly suggestive of esophageal perforation (Fig 1). The result of a CT scan of the abdomen was normal. A Gastrografin (Tyco/ Minimally Invasive Management MallinKrodt, St. Louis, MO) swallow demonstrated free of Boerhaave’s Syndrome extravasation of contrast from the left posterolateral Ahmad S. Ashrafi, MD, Omar Awais, DO, aspect of the distal esophagus just above the level of the and Miguel Alvelo-Rivera, MD hiatus (Fig 2). After aggressive volume resuscitation, commencement The Heart Lung and Esophageal Surgery Institute, University of Pittsburgh Medical Center, Pittsburgh, Pennsylvania of broad-spectrum antibiotics, and analgesia, the patient was taken to the operating room. On-table endoscopy revealed a 2-cm to 3-cm perforation just above a nar- We report the case of a 42-year-old man with Boerhaave’s rowed gastroesophageal junction. A laparoscopic explo- syndrome. His medical history was significant only for a ration showed no intraabdominal pathology. long-standing history of dysphagia. The patient pre- We then harvested a generous portion length of the Accepted for publication May 24, 2006. greater omentum and secured it to the edges of the left crus. We also performed a Heller myotomy given the Address correspondence to Dr Alvelo-Rivera, The Heart, Lung and Esophageal Surgery Institute, University of Pittsburgh Medical Center, patient’s long-standing history of dysphagia. A laparo- 200 Lothrop St, C-800, Pittsburgh, PA 15213; e-mail: alveloriveram@ scopic gastrostomy and feeding jejunostomy were per- upmc.edu. formed, and the port sites were closed. © 2007 by The Society of Thoracic Surgeons 0003-4975/07/$32.00 Published by Elsevier Inc doi:10.1016/j.athoracsur.2006.05.111
  • 2. 318 CASE REPORT ASHRAFI ET AL Ann Thorac Surg MINIMALLY INVASIVE MANAGEMENT OF BOERHAAVE’S 2007;83:317–9 Fig 1. Computed tomography image of the chest shows pneumome- diastinum with air tracking laterally towards the left pleural space and a left pleural effusion. FEATURE ARTICLES A double-lumen endotracheal tube was then inserted, and the patient was positioned in the right lateral decu- bitus position for video-assisted thoracoscopic explora- tion of the left chest. The esophageal perforation site was identified at a level just above the esophageal hiatus. A two-layer repair was performed using simple interrupted sutures. The harvested omentum was then used to cover the entire length of the repaired esophagus. Intraopera- tive insufflation of the esophagus did not show a leak, Fig 3. A postoperative barium swallow shows essentially a normal esophagus. and we placed two chest tubes and two Jackson-Pratt drains. Incisions were closed and dressings were applied. The patient was transferred to the intensive care unit and had an uneventful course. He was discharged to the ward the next day. A contrast study on postoperative day 4 showed no leak or obstruction (Fig 3). He was dis- charged home on postoperative day 9. Comment Boerhaave’s syndrome, or spontaneous (postemetic) per- foration of the esophagus, was first described by Her- mann Boerhaave in 1724 [1]. It is a very uncommon entity, with an estimated incidence in the literature of 1 in 6000 patients [2]. The esophagus differs from the rest of the alimentary tract in that it lacks a serosal layer, which normally contains collagen and elastic fibers. This makes it more susceptible to rupture at lower pressures than the rest of the gastrointestinal tract. Spontaneous esophageal rupture is well documented as a postemetic phenome- non. Early recognition and prompt treatment are impor- tant factors in minimizing the mortality. The mortality ranges from 20% to 30% [3], but if left untreated, ap- proaches 100%. Barrett reported the first case of success- ful surgical repair in 1947 [4]. Fig 2. A Gastrografin swallow shows extravasation of contrast in The minimally invasive technique could be used only if the left chest. the patient is hemodynamically stable, without signs of
  • 3. Ann Thorac Surg CASE REPORT ASHRAFI ET AL 319 2007;83:317–9 MINIMALLY INVASIVE MANAGEMENT OF BOERHAAVE’S escalating sepsis, without significant medical risk factors standpoint if the presentation is not acute, the perfora- that would preclude major surgery, and in patients with tion is well contained with good distal flow of contrast, no contraindications for laparoscopy or thoracoscopy. and in a patient with no signs of sepsis. In managing this patient, we began with laparoscopy to Postoperative morbidity includes the non-procedure- harvest the omentum, performed a gastrostomy, a feed- related postoperative complications, stricture formation, ing jejunostomy, and an esophagomyotomy. We also leak requiring further surgical management, or diver- wanted to assess potential intraabdominal extent of the sion/esophagectomy, with or without delayed injury. reconstruction. The repair was begun with a myotomy to identify the A Medline search of the literature pertaining to the true apices of the perforation. We then débrided the minimally invasive management of Boerhaave’s syn- nonviable tissue and performed an interrupted mucosal drome yielded two reports. The first describes a left repair by using absorbable suture material and a second thoracoscopic intracorporeal suture repair and drainage. layer of repair by approximating the esophageal muscle. The patient had developed a leak that was managed We then covered the repair with omentum. Other op- conservatively [5]. The second report describes a laparo- tions for buttressing include pleura, intercostal muscles, scopic primary repair and a 270° posterior fundoplication pericardial fat pad, or latissimus/serratus/pectoralis in a 72-year-old man. The patient was discharged home muscle. after 2 weeks of hospitalization with no leak [6]. Jackson-Pratt drains were used for management of Although open repair and drainage are the gold stan- potential postoperative leak to act as a controlled fistula, dard, we conclude that laparoscopic and thoracoscopic as this was a case of nonconventional surgical manage- management of Boerhaave’s syndrome is a feasible ment. It is possible to use alternative methods of drain- FEATURE ARTICLES alternative. age (eg, chest tube) or none at all, but we prefer to drain locally with Jackson-Pratt drains. Gastric decompression and nutritional support are References important aspects of the postoperative management. 1. Derbes VJ, Mitchell RE Jr. Herman Boerhaave Atrocis, nec Although a nasogastric tube is an alternative, we rou- descripti prius, morbi historia; the first translation of classic tinely perform laparoscopic jejunostomy and gastros- case report of rupture of the esophagus, with annotations. Bull Med Libr Assoc 1955;43:217–24. tomy tubes for other conditions, and usually it only adds 2. Lillington GA, Bernatz PE. Spontaneous perforation of esoph- 15 to 20 minutes to the operating time. This allows early agus. Dis Chest 1961;39:177– 84. institution of enteral feeding as well as a more secure way 3. Jougon J, Mc Bride T, Delcambre F, Minniti A, Velly JF. of keeping the stomach decompressed. Also, in the event Primary esophageal repair for Boerhaave’s syndrome what- of a prolonged course, it provides a more comfortable ever the free interval between perforation and treatment. Eur J Cardiothorac Surg 2004;25:475–9. drainage technique for the patient. Every patient under- 4. Barrett NR. Report of a case of spontaneous perforation of the going surgical management of Boerhaave’s syndrome esophagus successfully treated by operation. Br J Surg 1947; runs the risk of leak or delayed healing, or both. There- 35:216. fore, the feeding tube ensures optimal enteral nutrition 5. Scott HJ, Rosin RD. Thoracoscopic repair of a transmural in the event that the patient is not able to eat in the rupture of the oesophagus (Boerhaave’s syndrome). J R Soc Med 1995;88:414P–5P. postoperative period. 6. Landen S, El Nakadi I. Minimally invasive approach to We recommend conservative treatment to patients Boerhaave’s syndrome: a pilot study of three cases. Surg who would not tolerate an operation from a medical Endosc 2002;16:1354 –7.