Korean J Thorac Cardiovasc Surg 2014;47:560-562 ISSN: 2233-601X (Print)
□ Case Report □
http://dx.doi.org/10.5090/kjtcs.2014.47.6.560
ISSN: 2093-6516 (Online)
Pulmonary Vein to Esophageal Fistula after Staged Hybrid Totally Thoracoscopic Surgical and Percutaneous Radiofrequency Catheter Ablation: A Case Report Byung-Jo Park, M.D.1, Yong Han Kim, M.D.1, Dong Seop Jeong, M.D., Ph.D.1, Yong Soo Choi, M.D., Ph.D.1, Young Keun On, M.D., Ph.D.2
A case of a fistula running from the pulmonary vein to the esophagus after a staged hybrid procedure combining total thoracoscopic ablation and percutaneous radiofrequency catheter ablation has not been reported previously. We describe such a case in a 37-year-old man who was successfully treated by surgery. Key words: 1. Pulmonary veins 2. Esophageal fistula
computed tomography imaging revealed a collection of locu-
CASE REPORT
lated air measuring up to 2 cm in diameter between the right A 37-year-old man was admitted to our hospital with
side of the left atrium and the esophagus, with no definitive
long-standing, persistent, lone atrial fibrillation refractory to
evidence of a fistulous connection (Fig. 1). Brain computed
medical therapy and several attempts of cardioversion. The
tomography revealed an acute-onset left middle cerebral ar-
patient underwent a total thoracoscopic epicardial ablation
tery infarction. Emergency echocardiography revealed many
without any intraoperative or postoperative complications, fol-
floating microbubbles.
lowed by a percutaneous postprocedural electrophysiologic
An urgent surgical intervention was performed due to our
evaluation that included confirmation of pulmonary vein iso-
high level of suspicion for a left atrial or pulmonary ve-
lation and a cavotricuspid isthmus block on the tenth post-
nous-esophageal fistula. The right femoral area was prepared
operative day. He was discharged in sinus rhythm six days
for the possibility of an emergent cardiopulmonary bypass.
after the hybrid procedure.
The chest was opened through the fifth intercostal space via
Two weeks after discharge, the patient was admitted to the
a right posterolateral thoracotomy under one-lung ventilation
emergency room with right-sided motor weakness and par-
with a double lumen endotracheal tube. After anteriorly re-
esthesia of both upper and lower extremities. His fever reached
tracting the lung, a meticulous dissection of the posterior me-
40 C, and he had a white blood cell count of 17,690/ L. An
diastinum between the esophagus, right upper and lower pul-
electrocardiogram showed atrial fibrillation. Computed tomog-
monary veins, and left atrium was performed. A fistula 2 to
raphy imaging of the chest and brain was performed. Chest
3 mm in diameter was discovered between the right lower
o
1
2
Department of Thoracic and Cardiovascular Surgery and Division of Cardiology, Department of Internal Medicine, Samsung Medical Center, Sungkyunkwan University School of Medicine Received: June 3, 2014, Revised: August 17, 2014, Accepted: September 3, 2014, Published online: December 5, 2014 Corresponding author: Dong Seop Jeong, Department of Thoracic and Cardiovascular Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul 135-710, Korea (Tel) 82-2-3410-1278 (Fax) 82-2-3410-1680 (E-mail) [email protected] C The Korean Society for Thoracic and Cardiovascular Surgery. 2014. All right reserved. CC This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
− 560 −
Pulmonary Vein to Esophageal Fistula
Fig. 1. (A-C) Axial and (D-F) coronal views of computed tomography imaging show a loculated air density on the right side of the left atrium.
cided to carry out a primary repair of this defect because the defect site was small and relatively clear. The fistula was carefully debrided and repaired with intermittent 4-0 black silk sutures. The operation was successful and the postoperative course was uneventful. The patient’s rhythm was converted to sinus rhythm and sustained after cardioversion one day after surgery. The patient became alert on the third postoperative day, and was extubated on the fifth postoperative day. He was discharged 39 days postoperatively, with a motor power
Fig. 2. Intraoperative photograph of the fistula between the anterior surface of the esophagus and the right lower pulmonary vein after division.
score of three out of five on the Medical Research Council scale after rehabilitation therapy. On his last outpatient visit, two and a half months after surgery, motor power was fully recovered without any sequelae.
pulmonary vein and anterior surface of the esophagus. The
DISCUSSION
fistula was divided with Metzenbaum scissors (Fig. 2). Surprisingly, there was no bleeding from the fistula on the side of the right lower pulmonary vein. The internal orifice
A hybrid procedure of a total thoracoscopic epicardial abla-
was very small, with a diameter of approximately 1 mm. The
tion and percutaneous transvenous radiofrequency catheter
right lower pulmonary vein was repaired with a bovine peri-
ablation for lone atrial fibrillation, performed either simulta-
cardial patch, covered, and fixed with continuous poly-
neously as a single stage procedure or as a two-stage proce-
propylene sutures. After repairing the right lower pulmonary
dure, has been developed to overcome the limitations of each
vein, we consulted our hospital’s thoracic surgeon about re-
technique and result in better outcomes [1,2].
pairing the fistula opening on the esophageal side. He de-
The success rate of this therapy varies according to the
− 561 −
Byung-Jo Park, et al
specific goals of the intervention and the duration of atrial fi-
fistulas. A median sternotomy or a right or left thoracotomy
brillation (paroxysmal or persistent). Pison et al. [1] reported
with or without cardiopulmonary bypass is possible depend-
conversion to sinus rhythm after a single stage procedure
ing on the clinical situation and location of the fistula.
with a success rate of 83% after one year. Muneretto et al.
Surgical exposure in our case was achieved via a right post-
[2] reported conversion to sinus rhythm after a two-stage pro-
erolateral thoracotomy to expose the suspicious right pulmo-
cedure with a success rate of 91.6% at a mean follow-up of
nary veins and left atrium.
30 months. After being performed for the first time in South
In light of this rare but potentially lethal event, we suggest
Korea at the Samsung Medical Center in February 2012, we
the development of a better-defined consensus, based on fur-
have performed 50 staged hybrid procedures to date with a
ther studies, on whether hybrid thoracoscopic epicardial and
successful conversion to sinus rhythm in 48 out of 50
percutaneous catheter ablations should be performed simulta-
patients.
neously or as a staged procedure.
Unfortunately, we experienced a potentially fatal case of a fistula running from the pulmonary vein to the esophagus fis-
CONFLICT OF INTEREST
tula despite the high success rate of conversion to sinus rhythm. We believe our case of a right lower pulmonary venous-esophageal fistula after thoracoscopic epicardial and
No potential conflict of interest relevant to this article was reported.
transvenous catheter ablation to be the first reported in the literature. Even though the radiofrequency catheter ablation
REFERENCES
was performed after the total thoracoscopic epicardial ablation in a staged procedure, we think that there may have been some degree of infection or edema at the ablation sites due to the short (ten days) interval between the components of the hybrid procedure. The process of pulmonary vein isolation confirmation may injure this fragile tissue. The most commonly reported esophageal complication after radiofrequency catheter ablation are left atrial-esophageal fistulas, with an incidence rate of up to 0.2% [3]. These fistulas result from the usual anatomical relationship of the esophagus to the left atrium and pulmonary veins. However, unusual variations where the esophagus is situated rightward in the posterior mediastinum near the right pulmonary veins have been described [4], as in our present case. There is no definitive surgical approach for esophageal
1. Pison L, La Meir M, van Opstal J, Blaauw Y, Maessen J, Crijns HJ. Hybrid thoracoscopic surgical and transvenous catheter ablation of atrial fibrillation. J Am Coll Cardiol 2012;60:54-61. 2. Muneretto C, Bisleri G, Bontempi L, Curnis A. Durable staged hybrid ablation with thoracoscopic and percutaneous approach for treatment of long-standing atrial fibrillation: a 30-month assessment with continuous monitoring. J Thorac Cardiovasc Surg 2012;144:1460-5. 3. Dagres N, Hindricks G, Kottkamp H, et al. Complications of atrial fibrillation ablation in a high-volume center in 1,000 procedures: still cause for concern? J Cardiovasc Electrophysiol 2009;20:1014-9. 4. Cury RC, Abbara S, Schmidt S, et al. Relationship of the esophagus and aorta to the left atrium and pulmonary veins: implications for catheter ablation of atrial fibrillation. Heart Rhythm 2005;2:1317-23.
− 562 −
□ Case Report □
http://dx.doi.org/10.5090/kjtcs.2014.47.6.560
ISSN: 2093-6516 (Online)
Pulmonary Vein to Esophageal Fistula after Staged Hybrid Totally Thoracoscopic Surgical and Percutaneous Radiofrequency Catheter Ablation: A Case Report Byung-Jo Park, M.D.1, Yong Han Kim, M.D.1, Dong Seop Jeong, M.D., Ph.D.1, Yong Soo Choi, M.D., Ph.D.1, Young Keun On, M.D., Ph.D.2
A case of a fistula running from the pulmonary vein to the esophagus after a staged hybrid procedure combining total thoracoscopic ablation and percutaneous radiofrequency catheter ablation has not been reported previously. We describe such a case in a 37-year-old man who was successfully treated by surgery. Key words: 1. Pulmonary veins 2. Esophageal fistula
computed tomography imaging revealed a collection of locu-
CASE REPORT
lated air measuring up to 2 cm in diameter between the right A 37-year-old man was admitted to our hospital with
side of the left atrium and the esophagus, with no definitive
long-standing, persistent, lone atrial fibrillation refractory to
evidence of a fistulous connection (Fig. 1). Brain computed
medical therapy and several attempts of cardioversion. The
tomography revealed an acute-onset left middle cerebral ar-
patient underwent a total thoracoscopic epicardial ablation
tery infarction. Emergency echocardiography revealed many
without any intraoperative or postoperative complications, fol-
floating microbubbles.
lowed by a percutaneous postprocedural electrophysiologic
An urgent surgical intervention was performed due to our
evaluation that included confirmation of pulmonary vein iso-
high level of suspicion for a left atrial or pulmonary ve-
lation and a cavotricuspid isthmus block on the tenth post-
nous-esophageal fistula. The right femoral area was prepared
operative day. He was discharged in sinus rhythm six days
for the possibility of an emergent cardiopulmonary bypass.
after the hybrid procedure.
The chest was opened through the fifth intercostal space via
Two weeks after discharge, the patient was admitted to the
a right posterolateral thoracotomy under one-lung ventilation
emergency room with right-sided motor weakness and par-
with a double lumen endotracheal tube. After anteriorly re-
esthesia of both upper and lower extremities. His fever reached
tracting the lung, a meticulous dissection of the posterior me-
40 C, and he had a white blood cell count of 17,690/ L. An
diastinum between the esophagus, right upper and lower pul-
electrocardiogram showed atrial fibrillation. Computed tomog-
monary veins, and left atrium was performed. A fistula 2 to
raphy imaging of the chest and brain was performed. Chest
3 mm in diameter was discovered between the right lower
o
1
2
Department of Thoracic and Cardiovascular Surgery and Division of Cardiology, Department of Internal Medicine, Samsung Medical Center, Sungkyunkwan University School of Medicine Received: June 3, 2014, Revised: August 17, 2014, Accepted: September 3, 2014, Published online: December 5, 2014 Corresponding author: Dong Seop Jeong, Department of Thoracic and Cardiovascular Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul 135-710, Korea (Tel) 82-2-3410-1278 (Fax) 82-2-3410-1680 (E-mail) [email protected] C The Korean Society for Thoracic and Cardiovascular Surgery. 2014. All right reserved. CC This is an open access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
− 560 −
Pulmonary Vein to Esophageal Fistula
Fig. 1. (A-C) Axial and (D-F) coronal views of computed tomography imaging show a loculated air density on the right side of the left atrium.
cided to carry out a primary repair of this defect because the defect site was small and relatively clear. The fistula was carefully debrided and repaired with intermittent 4-0 black silk sutures. The operation was successful and the postoperative course was uneventful. The patient’s rhythm was converted to sinus rhythm and sustained after cardioversion one day after surgery. The patient became alert on the third postoperative day, and was extubated on the fifth postoperative day. He was discharged 39 days postoperatively, with a motor power
Fig. 2. Intraoperative photograph of the fistula between the anterior surface of the esophagus and the right lower pulmonary vein after division.
score of three out of five on the Medical Research Council scale after rehabilitation therapy. On his last outpatient visit, two and a half months after surgery, motor power was fully recovered without any sequelae.
pulmonary vein and anterior surface of the esophagus. The
DISCUSSION
fistula was divided with Metzenbaum scissors (Fig. 2). Surprisingly, there was no bleeding from the fistula on the side of the right lower pulmonary vein. The internal orifice
A hybrid procedure of a total thoracoscopic epicardial abla-
was very small, with a diameter of approximately 1 mm. The
tion and percutaneous transvenous radiofrequency catheter
right lower pulmonary vein was repaired with a bovine peri-
ablation for lone atrial fibrillation, performed either simulta-
cardial patch, covered, and fixed with continuous poly-
neously as a single stage procedure or as a two-stage proce-
propylene sutures. After repairing the right lower pulmonary
dure, has been developed to overcome the limitations of each
vein, we consulted our hospital’s thoracic surgeon about re-
technique and result in better outcomes [1,2].
pairing the fistula opening on the esophageal side. He de-
The success rate of this therapy varies according to the
− 561 −
Byung-Jo Park, et al
specific goals of the intervention and the duration of atrial fi-
fistulas. A median sternotomy or a right or left thoracotomy
brillation (paroxysmal or persistent). Pison et al. [1] reported
with or without cardiopulmonary bypass is possible depend-
conversion to sinus rhythm after a single stage procedure
ing on the clinical situation and location of the fistula.
with a success rate of 83% after one year. Muneretto et al.
Surgical exposure in our case was achieved via a right post-
[2] reported conversion to sinus rhythm after a two-stage pro-
erolateral thoracotomy to expose the suspicious right pulmo-
cedure with a success rate of 91.6% at a mean follow-up of
nary veins and left atrium.
30 months. After being performed for the first time in South
In light of this rare but potentially lethal event, we suggest
Korea at the Samsung Medical Center in February 2012, we
the development of a better-defined consensus, based on fur-
have performed 50 staged hybrid procedures to date with a
ther studies, on whether hybrid thoracoscopic epicardial and
successful conversion to sinus rhythm in 48 out of 50
percutaneous catheter ablations should be performed simulta-
patients.
neously or as a staged procedure.
Unfortunately, we experienced a potentially fatal case of a fistula running from the pulmonary vein to the esophagus fis-
CONFLICT OF INTEREST
tula despite the high success rate of conversion to sinus rhythm. We believe our case of a right lower pulmonary venous-esophageal fistula after thoracoscopic epicardial and
No potential conflict of interest relevant to this article was reported.
transvenous catheter ablation to be the first reported in the literature. Even though the radiofrequency catheter ablation
REFERENCES
was performed after the total thoracoscopic epicardial ablation in a staged procedure, we think that there may have been some degree of infection or edema at the ablation sites due to the short (ten days) interval between the components of the hybrid procedure. The process of pulmonary vein isolation confirmation may injure this fragile tissue. The most commonly reported esophageal complication after radiofrequency catheter ablation are left atrial-esophageal fistulas, with an incidence rate of up to 0.2% [3]. These fistulas result from the usual anatomical relationship of the esophagus to the left atrium and pulmonary veins. However, unusual variations where the esophagus is situated rightward in the posterior mediastinum near the right pulmonary veins have been described [4], as in our present case. There is no definitive surgical approach for esophageal
1. Pison L, La Meir M, van Opstal J, Blaauw Y, Maessen J, Crijns HJ. Hybrid thoracoscopic surgical and transvenous catheter ablation of atrial fibrillation. J Am Coll Cardiol 2012;60:54-61. 2. Muneretto C, Bisleri G, Bontempi L, Curnis A. Durable staged hybrid ablation with thoracoscopic and percutaneous approach for treatment of long-standing atrial fibrillation: a 30-month assessment with continuous monitoring. J Thorac Cardiovasc Surg 2012;144:1460-5. 3. Dagres N, Hindricks G, Kottkamp H, et al. Complications of atrial fibrillation ablation in a high-volume center in 1,000 procedures: still cause for concern? J Cardiovasc Electrophysiol 2009;20:1014-9. 4. Cury RC, Abbara S, Schmidt S, et al. Relationship of the esophagus and aorta to the left atrium and pulmonary veins: implications for catheter ablation of atrial fibrillation. Heart Rhythm 2005;2:1317-23.
− 562 −
