- Email: [email protected]
Successful Surgical Correction of Total Anomalous Pulmonary Venous Drainage in the Sixth Decade
1684
CASE REPORT MODI ET AL SURGICAL CORRECTION OF TAPVD
Ann Thorac Surg 2008;86:1684 – 6
Successful Surgical Correction of Total Anomalous Pulmonary Venous Drainage in the Sixth Decade Amit Modi, MRCS, Hunaid A. Vohra, MRCS, MD, Ivan Brown, FRCR, and Stephen M. Langley, FRCS(CTh) Wessex Cardiothoracic Centre, Southampton University Hospitals NHS Trust, Southampton, United Kingdom
We report an unusual case of total anomalous pulmonary venous drainage (TAPVD), who was successfully treated by surgery at the age of 56 years. We believe that this is the oldest person in the English literature to undergo surgical correction of TAPVD. The pathophysiology and factors for prolonged survival are further discussed. (Ann Thorac Surg 2008;86:1684 – 6) © 2008 by The Society of Thoracic Surgeons Fig 4. Pulmonary angiography after the hybrid transcatheter-surgical approach. (* ⫽ left pulmonary artery; ** ⫽ right pulmonary artery; MPA ⫽ main pulmonary artery.)
FEATURE ARTICLES
pathway and effectively break up the previously mentioned vicious circle. We believe that this approach has never been reported in literature and should always be kept in mind in these complex vascular obstructions, mainly in low-weight patients. However, only a close follow-up will disclose us the long-term outcome of these vessels, as well as the need of further surgical or percutaneous interventions as the infant grows. Nevertheless, the use of stents potentially re-dilatable up to the adult pulmonary artery size, as was in this patient, should allow additional nonsurgical dilatations whenever the clinical conditions warranted them.
References 1. Franceschini P, Guala A, Licata D, Di Cara G, Franceschini D. Arterial tortuosity sindrome. Am J Med Genet 2000;91: 141–3. 2. Zaidi SHE, Peltekova V, Meyer S, et al. A family exhibiting arterial tortuosity syndrome displays homozygosity for markers in the arterial tortuosity locus at chromosome 20q13. Clin Genet 2004;67:183– 8. 3. Hoop R, Steinmann B, Valsangiacomo Buechel ER, Cardiovascular findings in arterial tortuosity syndrome. Eur Heart J 2006;27:2045. 4. Bottio T, Bisleri G, Piccoli P, Muneretto C. Valve-sparing aortic root replacement in a patient with a rare connective tissue disorder: arterial tortuosity syndrome. J Thorac Cardiovasc Surg 2007;133:252–3. 5. Rivera IR, Gomes L, Moises VA, Silva CC, Andrade JL, Carvalho AC. Multiple arterial anomalies in the newborn infant. Echocardiographic and angiographic diagnosis. Arq Bras Cardiol 2000;75:141– 4. 6. Coucke PJ, Willaert A, Wessels MW, et al. Mutations in the facilitative glucose transported GLUT10 alter angiogenesis and cause arterial tortuosity sindrome. Nat Genet 2006;38: 452–7. © 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
T
otal anomalous pulmonary venous drainage (TAPVD) is rare, comprising of approximately 1.5% of all congenital heart abnormalities [1]. It is usually diagnosed in the neonatal period and is rarely seen in adults [2]. It requires surgical correction for survival as no catheter corrective treatment exists. In patients with TAPVD, all the venous blood returning from the lungs drains to the systemic veins, creating a large left-to-right shunt. Supply of oxygenated blood to the systemic circulation requires an atrial septal defect to allow oxygenated blood to enter the left ventricle. Without surgery, 80% babies with TAPVD die before their first birthday [3, 4]. We report a case of a 56-year-old woman who underwent successful surgical correction of TAPVD. The patient complained of breathlessness at 7 years of age during a ballet lesson. According to the patient, investigations performed at another center revealed anomalous pulmonary venous drainage, but it remained unknown whether it was partial or total. Cardiac catheterization confirmed the diagnosis but failed to identify the exact drainage pattern, which was diagnosed at the age of 41 years by magnetic resonance imaging scan. She refused surgery at that age due to quoted high risk. Eventually her symptoms of breathlessness deteriorated, and she presented to us as a new patient to our department at the age of 56 years. On examination, she had minimal cyanosis and early clubbing. Oxygen saturations were 85% on air, regular pulse of 90 per minute, blood pressure of 142/64 mm Hg, and an ejection systolic murmur at upper left sternal edge. Blood tests revealed
Accepted for publication April 25, 2008. Address correspondence to Dr Langley, Pediatric and Adult Cardiac Surgery, Pediatric Cardiac Surgical Research, Oregon Health and Science University, Doernbecher Children’s Hospital, Portland, OR 97239; e-mail: [email protected].
0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2008.04.091
Ann Thorac Surg 2008;86:1684 – 6
CASE REPORT MODI ET AL SURGICAL CORRECTION OF TAPVD
1685
transferred to the intensive care unit in a stable condition on no inotropes. The patient made an excellent recovery and the patient was discharged on postoperative day 7. At follow-up, 2 years post-surgery, she is in New York Heart Association functional class I. The transthoracic echocardiogram showed normal left ventricle function, good right ventricle function, thickened pulmonary valve, mean gradient of 23 mm Hg, dilated right heart, moderate tricuspid regurgitation, pulmonary veins draining normally to the left atrium, and no intracardiac shunt.
Fig 1. Cardiac magnetic resonance imaging (sagittal view) showing vertical vein (dotted white arrow) draining into the enlarged innominate vein (solid white arrow).
polycythaemia with hemoglobin of 182 gm/L and hematocrit of 51.5. An electrocardiogram demonstrated sinus rhythm with first degree heart block and a right bundle branch block. Chest roentgenogram revealed cardiomegaly with prominent superior vena cava and right heart enlargement. Transthoracic echocardiogram showed severely dilated right atrium and right ventricle, large atrial septal defect, and moderate tricuspid regurgitation (right ventricle systolic pressure ⫽ 90 mm Hg ⫹ right atrium pressure). The left ventricle was compressed by the right ventricle, but there was good left ventricle systolic function. The gradient across pulmonary valve was 50 mm Hg, the aortic arch was normal, and there was a pericardial effusion (⬍ 2 cm) with no tamponade. A cardiac magnetic resonance imaging scan (Magneton Avanto; Siemens, Malvern, PA) was repeated, which demonstrated supracardiac type of TAPVD (Fig 1) and a vertical vein draining into the innominate vein (Fig 2). Cardiac catheterization revealed supracardiac TAPVD, moderate pulmonary stenosis (peak gradient of 30 mm Hg across the pulmonary valve), Qp:Qs ⫽ 2.2:1, large atrial septal defect (shunting right to left), and normal coronary vessels. Surgical repair of TAPVD was carried out by a median sternotomy. The patient was placed on cardiopulmonary bypass by aorto-bi-caval cannulation and was cooled to 28° C. Antegrade cold blood cardioplegia was used. The left atrium was anastomosed to the pulmonary venous confluence and the atrial septal defect was partially closed leaving a small defect, and the vertical vein was ligated. The air was removed from the left heart and cardiopulmonary bypass was terminated. The crossclamp time was 67 minutes and the total cardiopulmonary bypass time was 167 minutes. The patient was
The TAPVD results from failure of primordial pulmonary vein to unite with the lung plexus of the veins. Therefore, the pulmonary veins drain to the heart through systemic veins to the right atrium. In TAPVD, supply of oxygenated blood to the systemic circulation requires intracardiac communication between the right and left sides of the heart to allow oxygenated blood to enter the left ventricle. The size of the communication determines the volume of blood able to cross to the left heart, and therefore it determines the cardiac output and systemic oxygenation. In supracardiac TAPVD, the ascending vein usually unites with the innominate vein, which may drain directly to the superior vena cava or the azygous vein. In the infracardiac TAPVD, a descending vein drains either to the inferior vena cava, the portal vein, hepatic veins, or ductus venosus. In cardiac TAPVD, the
Fig 2. Cardiac magnetic resonance imaging (sagittal view) showing right and left pulmonary veins (dotted white arrows) opening into the confluence (solid white arrow). Also seen are the ascending aorta above the pulmonary trunk, which is next to the vertical vein (grey arrow).
FEATURE ARTICLES
Comment
1686
CASE REPORT TERADA ET AL FOREIGN BODY EXTRACTED WITH TRANILAST
FEATURE ARTICLES
anomalous veins drain into the coronary sinus or directly to the right atrium. Patients with TAPVD usually present in the early neonatal period, often with profound cyanosis and shock, and they almost always require surgical treatment in the neonatal period [5, 6], often on an emergency basis. There are two anatomic factors that determine the patient’s clinical status. First, the patient’s cardiac output and supply of oxygenated blood is limited by the amount of blood that can cross the atrial septum. Therefore, the characteristics of the necessary right-to-left shunt determine systemic cardiac output and oxygenation. Second, an obstruction may occur in the path of the pulmonary venous drainage from the lungs to the systemic venous system. If obstruction occurs, egress of blood from the lungs is limited. The consequences of obstruction are limitation of pulmonary blood flow, pulmonary venous congestion, impairment of oxygenation, and elevation of pulmonary artery pressures. These events lead to lifethreatening cyanosis in neonates. Increased pulmonary blood flow and pulmonary venous obstruction will result in pulmonary hypertension and eventually heart failure. Survival until the age of 56 is unusual, and increasing age is described as an unfavorable factor for successful surgical outcome. In this particular patient, survival to this age can be attributed to supracardiac type of TAPVD, an unobstructed pulmonary venous flow, a large intracardiac shunt to maintain the cardiac output, and a normalsized left ventricle. Moreover, there were neither any associated anomalies nor any preoperative acidosis. Postrepair, there was no residual obstruction at the site of left atrial anastomosis, which could manifest as poor cardiac output and chest roentgenogram findings of pulmonary congestion. We believe that (on balance) the potential disadvantages of replacing the pulmonary valve (ie, increased length of surgery, long-term risks of valve complications, need for reoperation in the future) outweighed the advantages in a patient with a relatively small to moderate gradient across the valve. In fact, this decision probably proved to be correct as the patient is now 2 years postoperatively asymptomatic, with no increase in the gradient across the valve. Generally, longterm prognosis after successful repair of TAPVD is favorable. Approximately 10% to 15% of patients have evidence of late pulmonary vein obstruction, which tends to be recurrent and progressive. For this reason, longterm surveillance is important. Long-term follow-up showing freedom from events and death in patients undergoing surgery for TAPVD after 50 years of age remains to be determined. In conclusion, this report describes a case of an unobstructed supracardiac TAPVD in a 56-year-old woman who was successfully treated by surgery. At this age, surgery should not be excluded as an option. However, careful perioperative assessment of this small group of patients is needed to exclude the presence of anatomical and physiological factors that could affect the outcome adversely. For surgery to be successful, a clear management plan should be drawn up for the preoperative, operative, and postoperative management of such pa© 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Ann Thorac Surg 2008;86:1686 – 8
tients in consultation with pediatric cardiologists, cardiac surgeons, anesthetists, and intensivists. Despite an excellent early outcome, late outcome is unknown.
References 1. Ferencz C, Rubin JD, Loffredo CA, Magee CM. The epidemiology of congenital heart disease, the Baltimore-Washington infant study (1981–1989). Perspectives in pediatric cardiology, Vol 4. Mount Kisco, NY: Futura Publishing Co Inc; 1993. 2. Correa-Villasenor A, Ferencz C, Boughman JA, Neill CA. Total anomalous pulmonary venous return: familial and environmental factors. The Baltimore-Washington Infant Study Group. Teratology 1991;44:415–28. 3. Gathman GE, Nadas AS. Total anomalous pulmonary venous connection: clinical and physiologic observations of 75 pediatric patients. Circulation 1970;42:143–54. 4. Behrendt DM, Aberdeen E, Waterson DJ, Bonham-Carter RE. Total anomalous pulmonary venous drainage in infants. I. Clinical and hemodynamic findings, methods, and results of operation in 37 cases. Circulation 1972;46:347–56. 5. Sano S, Brawn WJ, Mee RB. Total anomalous pulmonary venous drainage. J Thorac Cardiovasc Surg 1989;97: 886 –92. 6. Yalta K, Turgut OO, Yilmaz A. Asymptomatic total anomalous pulmonary venous connection with double drainage in a young adult: a case report. Heart Surg Forum 1007;10: E211–2.
Intrabronchial Foreign Body Extracted With Tranilast and Corticosteroid Yasuji Terada, MD, Yasuto Sakaguchi, MD, Tomoya Kono, MD, Jun Nohara, MD, and Tetsuo Noguchi, MD Departments of Thoracic Surgery and Respiratory Medicine, Nagahama City Hospital, Nagahama, Japan
We present a case of intrabronchial foreign body buried in granulation tissue, which was successfully extracted administrating tranilast (n-[3,4-dimethoxycinnamoyl] anthranilic acid), suppressing collagen synthesis by fibroblasts in keloid and hypertrophic scars, and corticosteroid. Bronchoscopy of a 74-year-old man showed the nail was buried in reactive granulation tissue and could not be observed from the surface. Tranilast at 300 mg/day and methylprednisolone at 250 mg/day were prescribed for 4 days, followed by a reduction of the corticosteroid to 40 mg/day for 3 days. Seven days later, the granulation tissue and mucosal edema were diminished, and the nail was successfully extracted. (Ann Thorac Surg 2008;86:1686 – 8) © 2008 by The Society of Thoracic Surgeons
Accepted for publication April 16, 2008. Address correspondence to Dr Terada, Department of Thoracic Surgery, Nagahama City Hospital, 313 Oinui-cho, Nagahama, 526-8580, Japan; e-mail: [email protected].
0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2008.04.060
CASE REPORT MODI ET AL SURGICAL CORRECTION OF TAPVD
Ann Thorac Surg 2008;86:1684 – 6
Successful Surgical Correction of Total Anomalous Pulmonary Venous Drainage in the Sixth Decade Amit Modi, MRCS, Hunaid A. Vohra, MRCS, MD, Ivan Brown, FRCR, and Stephen M. Langley, FRCS(CTh) Wessex Cardiothoracic Centre, Southampton University Hospitals NHS Trust, Southampton, United Kingdom
We report an unusual case of total anomalous pulmonary venous drainage (TAPVD), who was successfully treated by surgery at the age of 56 years. We believe that this is the oldest person in the English literature to undergo surgical correction of TAPVD. The pathophysiology and factors for prolonged survival are further discussed. (Ann Thorac Surg 2008;86:1684 – 6) © 2008 by The Society of Thoracic Surgeons Fig 4. Pulmonary angiography after the hybrid transcatheter-surgical approach. (* ⫽ left pulmonary artery; ** ⫽ right pulmonary artery; MPA ⫽ main pulmonary artery.)
FEATURE ARTICLES
pathway and effectively break up the previously mentioned vicious circle. We believe that this approach has never been reported in literature and should always be kept in mind in these complex vascular obstructions, mainly in low-weight patients. However, only a close follow-up will disclose us the long-term outcome of these vessels, as well as the need of further surgical or percutaneous interventions as the infant grows. Nevertheless, the use of stents potentially re-dilatable up to the adult pulmonary artery size, as was in this patient, should allow additional nonsurgical dilatations whenever the clinical conditions warranted them.
References 1. Franceschini P, Guala A, Licata D, Di Cara G, Franceschini D. Arterial tortuosity sindrome. Am J Med Genet 2000;91: 141–3. 2. Zaidi SHE, Peltekova V, Meyer S, et al. A family exhibiting arterial tortuosity syndrome displays homozygosity for markers in the arterial tortuosity locus at chromosome 20q13. Clin Genet 2004;67:183– 8. 3. Hoop R, Steinmann B, Valsangiacomo Buechel ER, Cardiovascular findings in arterial tortuosity syndrome. Eur Heart J 2006;27:2045. 4. Bottio T, Bisleri G, Piccoli P, Muneretto C. Valve-sparing aortic root replacement in a patient with a rare connective tissue disorder: arterial tortuosity syndrome. J Thorac Cardiovasc Surg 2007;133:252–3. 5. Rivera IR, Gomes L, Moises VA, Silva CC, Andrade JL, Carvalho AC. Multiple arterial anomalies in the newborn infant. Echocardiographic and angiographic diagnosis. Arq Bras Cardiol 2000;75:141– 4. 6. Coucke PJ, Willaert A, Wessels MW, et al. Mutations in the facilitative glucose transported GLUT10 alter angiogenesis and cause arterial tortuosity sindrome. Nat Genet 2006;38: 452–7. © 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
T
otal anomalous pulmonary venous drainage (TAPVD) is rare, comprising of approximately 1.5% of all congenital heart abnormalities [1]. It is usually diagnosed in the neonatal period and is rarely seen in adults [2]. It requires surgical correction for survival as no catheter corrective treatment exists. In patients with TAPVD, all the venous blood returning from the lungs drains to the systemic veins, creating a large left-to-right shunt. Supply of oxygenated blood to the systemic circulation requires an atrial septal defect to allow oxygenated blood to enter the left ventricle. Without surgery, 80% babies with TAPVD die before their first birthday [3, 4]. We report a case of a 56-year-old woman who underwent successful surgical correction of TAPVD. The patient complained of breathlessness at 7 years of age during a ballet lesson. According to the patient, investigations performed at another center revealed anomalous pulmonary venous drainage, but it remained unknown whether it was partial or total. Cardiac catheterization confirmed the diagnosis but failed to identify the exact drainage pattern, which was diagnosed at the age of 41 years by magnetic resonance imaging scan. She refused surgery at that age due to quoted high risk. Eventually her symptoms of breathlessness deteriorated, and she presented to us as a new patient to our department at the age of 56 years. On examination, she had minimal cyanosis and early clubbing. Oxygen saturations were 85% on air, regular pulse of 90 per minute, blood pressure of 142/64 mm Hg, and an ejection systolic murmur at upper left sternal edge. Blood tests revealed
Accepted for publication April 25, 2008. Address correspondence to Dr Langley, Pediatric and Adult Cardiac Surgery, Pediatric Cardiac Surgical Research, Oregon Health and Science University, Doernbecher Children’s Hospital, Portland, OR 97239; e-mail: [email protected].
0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2008.04.091
Ann Thorac Surg 2008;86:1684 – 6
CASE REPORT MODI ET AL SURGICAL CORRECTION OF TAPVD
1685
transferred to the intensive care unit in a stable condition on no inotropes. The patient made an excellent recovery and the patient was discharged on postoperative day 7. At follow-up, 2 years post-surgery, she is in New York Heart Association functional class I. The transthoracic echocardiogram showed normal left ventricle function, good right ventricle function, thickened pulmonary valve, mean gradient of 23 mm Hg, dilated right heart, moderate tricuspid regurgitation, pulmonary veins draining normally to the left atrium, and no intracardiac shunt.
Fig 1. Cardiac magnetic resonance imaging (sagittal view) showing vertical vein (dotted white arrow) draining into the enlarged innominate vein (solid white arrow).
polycythaemia with hemoglobin of 182 gm/L and hematocrit of 51.5. An electrocardiogram demonstrated sinus rhythm with first degree heart block and a right bundle branch block. Chest roentgenogram revealed cardiomegaly with prominent superior vena cava and right heart enlargement. Transthoracic echocardiogram showed severely dilated right atrium and right ventricle, large atrial septal defect, and moderate tricuspid regurgitation (right ventricle systolic pressure ⫽ 90 mm Hg ⫹ right atrium pressure). The left ventricle was compressed by the right ventricle, but there was good left ventricle systolic function. The gradient across pulmonary valve was 50 mm Hg, the aortic arch was normal, and there was a pericardial effusion (⬍ 2 cm) with no tamponade. A cardiac magnetic resonance imaging scan (Magneton Avanto; Siemens, Malvern, PA) was repeated, which demonstrated supracardiac type of TAPVD (Fig 1) and a vertical vein draining into the innominate vein (Fig 2). Cardiac catheterization revealed supracardiac TAPVD, moderate pulmonary stenosis (peak gradient of 30 mm Hg across the pulmonary valve), Qp:Qs ⫽ 2.2:1, large atrial septal defect (shunting right to left), and normal coronary vessels. Surgical repair of TAPVD was carried out by a median sternotomy. The patient was placed on cardiopulmonary bypass by aorto-bi-caval cannulation and was cooled to 28° C. Antegrade cold blood cardioplegia was used. The left atrium was anastomosed to the pulmonary venous confluence and the atrial septal defect was partially closed leaving a small defect, and the vertical vein was ligated. The air was removed from the left heart and cardiopulmonary bypass was terminated. The crossclamp time was 67 minutes and the total cardiopulmonary bypass time was 167 minutes. The patient was
The TAPVD results from failure of primordial pulmonary vein to unite with the lung plexus of the veins. Therefore, the pulmonary veins drain to the heart through systemic veins to the right atrium. In TAPVD, supply of oxygenated blood to the systemic circulation requires intracardiac communication between the right and left sides of the heart to allow oxygenated blood to enter the left ventricle. The size of the communication determines the volume of blood able to cross to the left heart, and therefore it determines the cardiac output and systemic oxygenation. In supracardiac TAPVD, the ascending vein usually unites with the innominate vein, which may drain directly to the superior vena cava or the azygous vein. In the infracardiac TAPVD, a descending vein drains either to the inferior vena cava, the portal vein, hepatic veins, or ductus venosus. In cardiac TAPVD, the
Fig 2. Cardiac magnetic resonance imaging (sagittal view) showing right and left pulmonary veins (dotted white arrows) opening into the confluence (solid white arrow). Also seen are the ascending aorta above the pulmonary trunk, which is next to the vertical vein (grey arrow).
FEATURE ARTICLES
Comment
1686
CASE REPORT TERADA ET AL FOREIGN BODY EXTRACTED WITH TRANILAST
FEATURE ARTICLES
anomalous veins drain into the coronary sinus or directly to the right atrium. Patients with TAPVD usually present in the early neonatal period, often with profound cyanosis and shock, and they almost always require surgical treatment in the neonatal period [5, 6], often on an emergency basis. There are two anatomic factors that determine the patient’s clinical status. First, the patient’s cardiac output and supply of oxygenated blood is limited by the amount of blood that can cross the atrial septum. Therefore, the characteristics of the necessary right-to-left shunt determine systemic cardiac output and oxygenation. Second, an obstruction may occur in the path of the pulmonary venous drainage from the lungs to the systemic venous system. If obstruction occurs, egress of blood from the lungs is limited. The consequences of obstruction are limitation of pulmonary blood flow, pulmonary venous congestion, impairment of oxygenation, and elevation of pulmonary artery pressures. These events lead to lifethreatening cyanosis in neonates. Increased pulmonary blood flow and pulmonary venous obstruction will result in pulmonary hypertension and eventually heart failure. Survival until the age of 56 is unusual, and increasing age is described as an unfavorable factor for successful surgical outcome. In this particular patient, survival to this age can be attributed to supracardiac type of TAPVD, an unobstructed pulmonary venous flow, a large intracardiac shunt to maintain the cardiac output, and a normalsized left ventricle. Moreover, there were neither any associated anomalies nor any preoperative acidosis. Postrepair, there was no residual obstruction at the site of left atrial anastomosis, which could manifest as poor cardiac output and chest roentgenogram findings of pulmonary congestion. We believe that (on balance) the potential disadvantages of replacing the pulmonary valve (ie, increased length of surgery, long-term risks of valve complications, need for reoperation in the future) outweighed the advantages in a patient with a relatively small to moderate gradient across the valve. In fact, this decision probably proved to be correct as the patient is now 2 years postoperatively asymptomatic, with no increase in the gradient across the valve. Generally, longterm prognosis after successful repair of TAPVD is favorable. Approximately 10% to 15% of patients have evidence of late pulmonary vein obstruction, which tends to be recurrent and progressive. For this reason, longterm surveillance is important. Long-term follow-up showing freedom from events and death in patients undergoing surgery for TAPVD after 50 years of age remains to be determined. In conclusion, this report describes a case of an unobstructed supracardiac TAPVD in a 56-year-old woman who was successfully treated by surgery. At this age, surgery should not be excluded as an option. However, careful perioperative assessment of this small group of patients is needed to exclude the presence of anatomical and physiological factors that could affect the outcome adversely. For surgery to be successful, a clear management plan should be drawn up for the preoperative, operative, and postoperative management of such pa© 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Ann Thorac Surg 2008;86:1686 – 8
tients in consultation with pediatric cardiologists, cardiac surgeons, anesthetists, and intensivists. Despite an excellent early outcome, late outcome is unknown.
References 1. Ferencz C, Rubin JD, Loffredo CA, Magee CM. The epidemiology of congenital heart disease, the Baltimore-Washington infant study (1981–1989). Perspectives in pediatric cardiology, Vol 4. Mount Kisco, NY: Futura Publishing Co Inc; 1993. 2. Correa-Villasenor A, Ferencz C, Boughman JA, Neill CA. Total anomalous pulmonary venous return: familial and environmental factors. The Baltimore-Washington Infant Study Group. Teratology 1991;44:415–28. 3. Gathman GE, Nadas AS. Total anomalous pulmonary venous connection: clinical and physiologic observations of 75 pediatric patients. Circulation 1970;42:143–54. 4. Behrendt DM, Aberdeen E, Waterson DJ, Bonham-Carter RE. Total anomalous pulmonary venous drainage in infants. I. Clinical and hemodynamic findings, methods, and results of operation in 37 cases. Circulation 1972;46:347–56. 5. Sano S, Brawn WJ, Mee RB. Total anomalous pulmonary venous drainage. J Thorac Cardiovasc Surg 1989;97: 886 –92. 6. Yalta K, Turgut OO, Yilmaz A. Asymptomatic total anomalous pulmonary venous connection with double drainage in a young adult: a case report. Heart Surg Forum 1007;10: E211–2.
Intrabronchial Foreign Body Extracted With Tranilast and Corticosteroid Yasuji Terada, MD, Yasuto Sakaguchi, MD, Tomoya Kono, MD, Jun Nohara, MD, and Tetsuo Noguchi, MD Departments of Thoracic Surgery and Respiratory Medicine, Nagahama City Hospital, Nagahama, Japan
We present a case of intrabronchial foreign body buried in granulation tissue, which was successfully extracted administrating tranilast (n-[3,4-dimethoxycinnamoyl] anthranilic acid), suppressing collagen synthesis by fibroblasts in keloid and hypertrophic scars, and corticosteroid. Bronchoscopy of a 74-year-old man showed the nail was buried in reactive granulation tissue and could not be observed from the surface. Tranilast at 300 mg/day and methylprednisolone at 250 mg/day were prescribed for 4 days, followed by a reduction of the corticosteroid to 40 mg/day for 3 days. Seven days later, the granulation tissue and mucosal edema were diminished, and the nail was successfully extracted. (Ann Thorac Surg 2008;86:1686 – 8) © 2008 by The Society of Thoracic Surgeons
Accepted for publication April 16, 2008. Address correspondence to Dr Terada, Department of Thoracic Surgery, Nagahama City Hospital, 313 Oinui-cho, Nagahama, 526-8580, Japan; e-mail: [email protected].
0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2008.04.060