- Email: [email protected]
Presentation of Pulmonary Artery Intimal Sarcoma in an Infant With a History of Neonatal Valvular Pulmonic Stenosis
1092
CASE REPORT CHAPPELL ET AL PULMONARY ARTERY INTIMAL SARCOMA IN INFANT
restrictive. This differs from most forms of infracardiac TAPVC in which patients present early and often unwell with cyanosis and obstruction to the pulmonary venous pathway. In TAPVC to the supradiaphragmatic IVC, the venous pathway does not pass through the diaphragm, hepatic parenchyma, or closing ductus venosus, all frequent positions of obstruction. Our experience is similar to other reports [1, 4, 5], including one of a 12-year-old presenting for the first time with TAPVC to the IVC [1]. In summary, TAPVC to the supradiaphragmatic IVC is a rare diagnosis and can masquerade as cardiac TAPVC. It is of diagnostic importance to identify the pulmonary venous confluence, coronary sinus, right atrium, and direct connections between these structures to prevent misdiagnosis. If there is any further diagnostic uncertainty, threedimensional imaging is recommended as surgical repair of TAPVC to IVC differs with that for cardiac TAPVC.
FEATURE ARTICLES
We would like to thank Joanne Wolfenden and Manjit Jonsen for assistance with imaging and Emily McIntosh for artwork. Dr Anna Seale is a research fellow at the Royal Brompton Hospital supported by grants from the Harrison Heart Foundation and the Joe Gandon Memorial Trust.
References 1. Wang ZP, Zhong F, Huang LB, Zhao W, Huang WM. Total anomalous pulmonary venous draining to inferior vena cava. Thorac Cardiov Surg 2003;51:226 – 8. 2. Duff DF, Nihill MR, McNamara DG. Infracardiac total anomalous pulmonary venous return. Review of clinical and pathological findings and results of operation in 28 cases. Br Heart J 1977;39:619 –26. 3. Freedom RM, Mawson JB, Yoo SJ, Benson LN. Abnormalities of pulmonary venous connections including subdivided left atrium. Congenital heart disease: textbook of angiography. Armonk, NY: Futura Publishing, 1997. 4. VanSon JAM, Hambsch J, Kinzel P, Haas GS, Mohr FW. Urgency of operation in infracardiac total anomalous pulmonary venous connection. Ann Thorac Surg 2000;70:128 –30. 5. Duff DF, Nihill MR, Vargo TA, Cooley DA. Infradiaphragmatic total anomalous pulmonary venous return: diagnosis and surgical repair in a 10-year-old child. Br Heart J 1975;37:1093– 6.
Presentation of Pulmonary Artery Intimal Sarcoma in an Infant With a History of Neonatal Valvular Pulmonic Stenosis Tresa Chappell, MD, C. Buddy Creech, MD, MPH, David Parra, MD, Arnold Strauss, MD, Frank Scholl, MD, and Gina Whitney, MD Department of Pediatrics, Divisions of Infectious Diseases, Cardiology, and Critical Care Medicine, and Department of Cardiothoracic Surgery, Vanderbilt Children’s Hospital, Nashville, Tennessee
Intimal sarcoma of the pulmonary artery is rare in the adult population. It is usually diagnosed postmortem in Accepted for publication Aug 21, 2007. Address correspondence to Dr Whitney, 2200 Children’s Way, 5121DOT, Nashville, TN 37232; e-mail: [email protected].
© 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Ann Thorac Surg 2008;85:1092– 4
patients thought to have pulmonary emboli. We present a case of intimal sarcoma of the pulmonary artery in an infant with a history of neonatal pulmonic stenosis. (Ann Thorac Surg 2008;85:1092– 4) © 2008 by The Society of Thoracic Surgeons
P
ulmonary artery sarcoma is a rare tumor described in the adult literature that often presents with pulmonary hypertension and right ventricular dysfunction. Patients are often mistakenly thought to have pulmonary thromboemboli, although advances in cardiac magnetic resonance imaging (MRI) have resulted in improved diagnostic accuracy. The diagnosis of pulmonary artery sarcoma carried a poor prognosis, although combined surgical and chemotherapuetic management offers the best hope of survival. A 2-month-old male presented with tachypnea, poor feeding, and a heart murmur. He was found to have moderate supravalvar and valvar pulmonic stenosis with a peak systolic gradient of 55 mm Hg. He underwent balloon valvuloplasty without relief of the right ventricular outflow tract (RVOT) obstruction. He then underwent pulmonary patch arterioplasty and partial valvotomy, during which the valve was found to be thickened. Owing to residual RVOT obstruction, he underwent resection of the left anterior leaflet. Intraoperative transesophageal echocardiograhy showed no residual RVOT obstruction and moderate pulmonary insufficiency. He did well and was discharged home 4 days postoperatively. Over the next 2 months, the patient continued to have failure to thrive. At 4 months of age, the patient presented with increased work of breathing and poor feeding. A chest radiograph showed cardiomegaly, bilateral pulmonary infiltrates, and a small right-side pleural effusion. Echocardiogram revealed severe pulmonary stenosis with a peak gradient of 100 mm Hg and a 17 ⫻ 10 mm mass extending from the pulmonary valve to the pulmonary artery bifurcation (Fig 1). He underwent emergent resection of the pulmonary valve and pulmonary artery mass. He was treated with vancomycin and gentamicin for presumed endocarditis. On postoperative day 28, after multiple failed attempts at ventilator weaning and continued fevers despite broad spectrum antibiotics, chest computed tomography scan showed unchanged pulmonary nodules and new mass lesions in the ribs and thoracic spine. The etiology of these lesions was thought to be infectious in nature, despite an extensive negative work-up to that point. A biopsy of a chest wall lesion was obtained. The tissue obtained showed unclassified pleomorphic spindle cell sarcoma with features of intimal sarcoma. Review of the resected pulmonary artery “thrombus” showed spindle cells and other similar histologic features. Repeat echocardiogram at that time showed regrowth of the main pulmonary artery mass. Chemotherapy with cytoxan, doxycycline, and vincristine alternating with etoposide and ifosfamide was initiated. A cardiac MRI scan was obtained at the beginning of therapy and 0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2007.08.072
Fig 1. Echocardiogram in the parasternal short axis view. An echo-bright mass was noted in the main pulmonary artery (MPA), extending into the left pulmonary artery (LPA). The right pulmonary artery (RPA) appears widely patent.
showed the mass in the RVOT blocking the branching of the left pulmonary artery (Fig 2). After several months of chemotherapy, the patient’s ventilatory status slowly improved, and he was able to transition to a home ventilator, allowing him to be discharged home between treatments. A repeat cardiac MRI scan 3 months into chemotherapy showed significant decrease in the size of the RVOT mass (Fig 3), and subsequent computed tomography scans have shown a decrease in tumor mass in his thoracic spine and lungs.
Comment Pulmonary arterial sarcomas are exceedingly rare and most often diagnosed in adult patients. Since the initial description in 1923 by Mandelstamm, approximately 200 cases in patients aged 13 to 81 years, with an average age at presentation of 49.3 years [1], have been reported. Farooki and coworkers [2] reported 2 cases of pulmonary artery sarcoma, both in teenagers, but both cases were consistent with hemangiopericytoma upon pathology review. Therefore, this patient is the first report of pulmo-
Fig 2. Cardiac magnetic resonance imaging axial double inversion recovery fast spin echo image showing the mass in the main pulmonary artery (MPA), extending into the left pulmonary artery (LPA). The right pulmonary artery (RPA) appears widely patent.
CASE REPORT CHAPPELL ET AL PULMONARY ARTERY INTIMAL SARCOMA IN INFANT
1093
Fig 3. Cardiac magnetic resonance imaging axial double inversion recovery fast spin echo image obtained 3 months into chemotherapy shows significant decrease in the size of the mass with no obstruction in the main pulmonary artery (MPA), left pulmonary artery (LPA), or right pulmonary artery (RPA).
nary artery sarcoma in an infant and of intimal sarcoma in a child. Patients with pulmonary arterial tumors usually present with evidence of pulmonary arterial hypertension and right ventricular failure. As would be expected, patients are often misdiagnosed with pulmonary thromboemboli, as was the case with this infant. On review of 100 cases by Nonomura and coworkers [3], only 7 patients were diagnosed preoperatively or antemortem. At the time of excision, the tumors may be mistaken for mucoid-appearing thrombi within the lumen of the pulmonary artery. Pathology examination reveals tissue of intimal origin, with growth into the lumen of the vessel. Most are poorly differentiated or undifferentiated. The true incidence of pulmonary arterial tumors is likely underestimated because of the misdiagnosis of thromboembolic disease. Findings such as fever, weight loss, and anemia may help distinguish pulmonary arterial malignancy from obstruction due to congenital or thromboembolic disease [1]. Echocardiography may allow visualization of the tumor within the right ventricular outflow tract. Advances in cardiac MRI imaging have improved the rate of preoperative diagnosis substantially in recent years. Factors that favor diagnosis of pulmonary artery sarcoma are heterogeneous soft tissue density, vascular distension, and enhancement of the gadolinium contrast [1]. Although pulmonary artery sarcoma carries a poor prognosis because of difficulty in diagnosis and high metastatic potential, surgical resection offers the best chance of survival. In a review of 93 cases of pulmonary artery sarcoma, Kruger and associates [4] reported a median survival of 1.5 months after diagnosis. However, the 27 patients who underwent surgical resection at the time of diagnosis survived an average of 10 months. The role of chemotherapy in the treatment of pulmonary artery sarcoma is unclear. A recent review reported about a 50% response rate to palliative chemotherapy with anthracyclines and ifosfamide in patients with advanced pulmonary artery sarcoma [5]. There is little information to direct treatment or predict outcome in this patient, given the rarity of this tumor in
FEATURE ARTICLES
Ann Thorac Surg 2008;85:1092– 4
1094
CASE REPORT KADOHAMA ET AL VAC FOR PEDIATRIC MEDIASTINITIS
children. His tumor burden has steadily decreased with surgery and chemotherapy. Given the experience in adults, his long-term prognosis is poor. It is likely that his original pulmonic stenosis at 2 months of age was due to tumor growth at the valve. Therefore, pulmonary artery sarcoma should be considered in the differential diagnosis of pulmonary stenosis presenting in infancy.
References
FEATURE ARTICLES
1. Yi ES. Tumors of the pulmonary vasculature. Cardiol Clin 2004;22:431– 40. 2. Farooki Z, Chang C, Jackson WL, et al. Primary pulmonary artery sarcoma in two children. Pediatr Cardiol 1988;9:243–51. 3. Nonomura A, Kurumaya H, Kono N, et al. Report of two autopsy cases studied by immunohistochemistry and electron microscopy and review of 110 cases reported in the literature. Acta Pathol Jpn 1988;38:883–96. 4. Kruger I, Borowski A, Horst M, de Vivie ER, Theissen P, Gross-Fengels W. Symptoms, diagnosis, and therapy of pulmonary sarcomas of the pulmonary artery. Thorac Cardiovasc Surg 1990;38:91–5. 5. Manso L, Alvarez E, Quintela M, Cortes-Funes H, Hitt R. Primary pulmonary artery sarcoma: report of three cases and review of the literature. Clin Lung Cancer 2007;8:277– 81.
Vacuum-Assisted Closure for Pediatric Post-Sternotomy Mediastinitis: Are Low Negative Pressures Sufficient? Takayuki Kadohama, MD, PhD, Nobuyuki Akasaka, MD, PhD, Akira Nagamine, MD, Keisuke Nakanishi, MD, Keiko Kiyokawa, MD, Kazutomo Goh, MD, PhD, and Tadahiro Sasajima, MD, PhD Departments of Surgery and Emergency Medicine, Asahikawa Medical University, Asahikawa, Japan
We present 3 cases of pediatric post-sternotomy mediastinitis treated by a vacuum-assisted closure (VAC). The patients 2 girls, aged 6 months and 10 months, and a 2-year-old boy. The onset of infection was at 9, 14, and 32 postoperative days. The culture examination detected coagulase-negative Staphylococci strains in 2 cases, and Staphylococcus aureus in 1 case. A VAC was performed at ⴚ50 mm Hg for 10, 12, and 7 days. The wounds were closed without vascularized soft tissue. A VAC under a low negative pressure is a useful and safe procedure for the management of pediatric post-sternotomy mediastinitis. (Ann Thorac Surg 2008;85:1094 – 6) © 2008 by The Society of Thoracic Surgeons Accepted for publication Sept 4, 2007. Address correspondence to Dr Kadohama, Asahikawa Medical University, Department of Surgery, Midorigaoka-Higashi 2-1-1-1, Asahikawa, 078-8510, Japan; e-mail: [email protected].
© 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Ann Thorac Surg 2008;85:1094 – 6
P
ost-sternotomy mediastinitis, also commonly called deep sternal wound infection, is one of the most feared complications in patients undergoing cardiac surgery [1]. At the present time, there is no general consensus regarding the appropriate surgical approach to mediastinitis after cardiac surgery. Vacuum-assisted closure (VAC) is a novel treatment with an ingenious mechanism, and recent publications have demonstrated encouraging clinical results [2]. However, the validity of VAC therapy for managing pediatric post-sternotomy mediastinitis remains to be confirmed because there have been very few reports published on this topic [3, 4]. We herein describe 3 cases of pediatric post-sternotomy mediastinitis treated by VAC therapy. Patient 1 was a 6-month-old girl who underwent a bilateral bidirectional Glenn shunt procedure for a tricuspid atresia with a concomitant pulmonary atresia. The surgery was performed urgently owing to clinical deterioration, as evidenced by the progression of cyanosis, which required intubation and mechanical control of ventilation. Nine days after surgery, she was noted to have a purulent discharge from the median sternal wound and a high-grade fever. Removal of the sternal wires and surgical debridement were performed. Thereafter, polyurethane foam, which was shaped to fit the defect, was placed within the cavity. The area was covered with an adhesive drape, and suction drainage was carried out at ⫺50 mm Hg for 10 days in addition to the administration of appropriate antibiotics. Coagulasenegative Staphylococci strains were detected by the culture examination. The polyurethane foam was replaced every few days. The VAC therapy lasted for 10 days. The wound was closed without any surgical revisions at 3 days after the discontinuation of VAC (Fig 1). Patient 2 was a 2-year old boy who underwent a pacemaker implantation using myocardial leads for a sick sinus syndrome. A total anomalous pulmonary venous connection repair and a release of a pulmonary venous obstruction were performed twice before this operation. The patient was noted to have a high-grade fever and a purulent discharge from the median sternal wound 14 days after surgery. Coagulase-negative Staphylococci strains were detected by the culture examination. Debridement of infected tissue was performed at the time of VAC application. We employed VAC therapy for 12 days until the wound became smaller and granulation tissue proliferated, in addition to the administration of appropriate antibiotics. However, there was still a small defect in the epithelialization. Finally, a second primary closure was performed at 4 weeks after VAC application. Patient 3 was a 10-month old girl who underwent a ventricular septal defect (VSD) repair. After she was discharged from the hospital (postoperative day 32), she was noted to have a high-grade fever and a purulent discharge from the median sternal wound. Staphylococcus aureus was detected by the culture examination. Debridement of the infected tissue was performed at the time of 0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2007.09.004
CASE REPORT CHAPPELL ET AL PULMONARY ARTERY INTIMAL SARCOMA IN INFANT
restrictive. This differs from most forms of infracardiac TAPVC in which patients present early and often unwell with cyanosis and obstruction to the pulmonary venous pathway. In TAPVC to the supradiaphragmatic IVC, the venous pathway does not pass through the diaphragm, hepatic parenchyma, or closing ductus venosus, all frequent positions of obstruction. Our experience is similar to other reports [1, 4, 5], including one of a 12-year-old presenting for the first time with TAPVC to the IVC [1]. In summary, TAPVC to the supradiaphragmatic IVC is a rare diagnosis and can masquerade as cardiac TAPVC. It is of diagnostic importance to identify the pulmonary venous confluence, coronary sinus, right atrium, and direct connections between these structures to prevent misdiagnosis. If there is any further diagnostic uncertainty, threedimensional imaging is recommended as surgical repair of TAPVC to IVC differs with that for cardiac TAPVC.
FEATURE ARTICLES
We would like to thank Joanne Wolfenden and Manjit Jonsen for assistance with imaging and Emily McIntosh for artwork. Dr Anna Seale is a research fellow at the Royal Brompton Hospital supported by grants from the Harrison Heart Foundation and the Joe Gandon Memorial Trust.
References 1. Wang ZP, Zhong F, Huang LB, Zhao W, Huang WM. Total anomalous pulmonary venous draining to inferior vena cava. Thorac Cardiov Surg 2003;51:226 – 8. 2. Duff DF, Nihill MR, McNamara DG. Infracardiac total anomalous pulmonary venous return. Review of clinical and pathological findings and results of operation in 28 cases. Br Heart J 1977;39:619 –26. 3. Freedom RM, Mawson JB, Yoo SJ, Benson LN. Abnormalities of pulmonary venous connections including subdivided left atrium. Congenital heart disease: textbook of angiography. Armonk, NY: Futura Publishing, 1997. 4. VanSon JAM, Hambsch J, Kinzel P, Haas GS, Mohr FW. Urgency of operation in infracardiac total anomalous pulmonary venous connection. Ann Thorac Surg 2000;70:128 –30. 5. Duff DF, Nihill MR, Vargo TA, Cooley DA. Infradiaphragmatic total anomalous pulmonary venous return: diagnosis and surgical repair in a 10-year-old child. Br Heart J 1975;37:1093– 6.
Presentation of Pulmonary Artery Intimal Sarcoma in an Infant With a History of Neonatal Valvular Pulmonic Stenosis Tresa Chappell, MD, C. Buddy Creech, MD, MPH, David Parra, MD, Arnold Strauss, MD, Frank Scholl, MD, and Gina Whitney, MD Department of Pediatrics, Divisions of Infectious Diseases, Cardiology, and Critical Care Medicine, and Department of Cardiothoracic Surgery, Vanderbilt Children’s Hospital, Nashville, Tennessee
Intimal sarcoma of the pulmonary artery is rare in the adult population. It is usually diagnosed postmortem in Accepted for publication Aug 21, 2007. Address correspondence to Dr Whitney, 2200 Children’s Way, 5121DOT, Nashville, TN 37232; e-mail: [email protected].
© 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Ann Thorac Surg 2008;85:1092– 4
patients thought to have pulmonary emboli. We present a case of intimal sarcoma of the pulmonary artery in an infant with a history of neonatal pulmonic stenosis. (Ann Thorac Surg 2008;85:1092– 4) © 2008 by The Society of Thoracic Surgeons
P
ulmonary artery sarcoma is a rare tumor described in the adult literature that often presents with pulmonary hypertension and right ventricular dysfunction. Patients are often mistakenly thought to have pulmonary thromboemboli, although advances in cardiac magnetic resonance imaging (MRI) have resulted in improved diagnostic accuracy. The diagnosis of pulmonary artery sarcoma carried a poor prognosis, although combined surgical and chemotherapuetic management offers the best hope of survival. A 2-month-old male presented with tachypnea, poor feeding, and a heart murmur. He was found to have moderate supravalvar and valvar pulmonic stenosis with a peak systolic gradient of 55 mm Hg. He underwent balloon valvuloplasty without relief of the right ventricular outflow tract (RVOT) obstruction. He then underwent pulmonary patch arterioplasty and partial valvotomy, during which the valve was found to be thickened. Owing to residual RVOT obstruction, he underwent resection of the left anterior leaflet. Intraoperative transesophageal echocardiograhy showed no residual RVOT obstruction and moderate pulmonary insufficiency. He did well and was discharged home 4 days postoperatively. Over the next 2 months, the patient continued to have failure to thrive. At 4 months of age, the patient presented with increased work of breathing and poor feeding. A chest radiograph showed cardiomegaly, bilateral pulmonary infiltrates, and a small right-side pleural effusion. Echocardiogram revealed severe pulmonary stenosis with a peak gradient of 100 mm Hg and a 17 ⫻ 10 mm mass extending from the pulmonary valve to the pulmonary artery bifurcation (Fig 1). He underwent emergent resection of the pulmonary valve and pulmonary artery mass. He was treated with vancomycin and gentamicin for presumed endocarditis. On postoperative day 28, after multiple failed attempts at ventilator weaning and continued fevers despite broad spectrum antibiotics, chest computed tomography scan showed unchanged pulmonary nodules and new mass lesions in the ribs and thoracic spine. The etiology of these lesions was thought to be infectious in nature, despite an extensive negative work-up to that point. A biopsy of a chest wall lesion was obtained. The tissue obtained showed unclassified pleomorphic spindle cell sarcoma with features of intimal sarcoma. Review of the resected pulmonary artery “thrombus” showed spindle cells and other similar histologic features. Repeat echocardiogram at that time showed regrowth of the main pulmonary artery mass. Chemotherapy with cytoxan, doxycycline, and vincristine alternating with etoposide and ifosfamide was initiated. A cardiac MRI scan was obtained at the beginning of therapy and 0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2007.08.072
Fig 1. Echocardiogram in the parasternal short axis view. An echo-bright mass was noted in the main pulmonary artery (MPA), extending into the left pulmonary artery (LPA). The right pulmonary artery (RPA) appears widely patent.
showed the mass in the RVOT blocking the branching of the left pulmonary artery (Fig 2). After several months of chemotherapy, the patient’s ventilatory status slowly improved, and he was able to transition to a home ventilator, allowing him to be discharged home between treatments. A repeat cardiac MRI scan 3 months into chemotherapy showed significant decrease in the size of the RVOT mass (Fig 3), and subsequent computed tomography scans have shown a decrease in tumor mass in his thoracic spine and lungs.
Comment Pulmonary arterial sarcomas are exceedingly rare and most often diagnosed in adult patients. Since the initial description in 1923 by Mandelstamm, approximately 200 cases in patients aged 13 to 81 years, with an average age at presentation of 49.3 years [1], have been reported. Farooki and coworkers [2] reported 2 cases of pulmonary artery sarcoma, both in teenagers, but both cases were consistent with hemangiopericytoma upon pathology review. Therefore, this patient is the first report of pulmo-
Fig 2. Cardiac magnetic resonance imaging axial double inversion recovery fast spin echo image showing the mass in the main pulmonary artery (MPA), extending into the left pulmonary artery (LPA). The right pulmonary artery (RPA) appears widely patent.
CASE REPORT CHAPPELL ET AL PULMONARY ARTERY INTIMAL SARCOMA IN INFANT
1093
Fig 3. Cardiac magnetic resonance imaging axial double inversion recovery fast spin echo image obtained 3 months into chemotherapy shows significant decrease in the size of the mass with no obstruction in the main pulmonary artery (MPA), left pulmonary artery (LPA), or right pulmonary artery (RPA).
nary artery sarcoma in an infant and of intimal sarcoma in a child. Patients with pulmonary arterial tumors usually present with evidence of pulmonary arterial hypertension and right ventricular failure. As would be expected, patients are often misdiagnosed with pulmonary thromboemboli, as was the case with this infant. On review of 100 cases by Nonomura and coworkers [3], only 7 patients were diagnosed preoperatively or antemortem. At the time of excision, the tumors may be mistaken for mucoid-appearing thrombi within the lumen of the pulmonary artery. Pathology examination reveals tissue of intimal origin, with growth into the lumen of the vessel. Most are poorly differentiated or undifferentiated. The true incidence of pulmonary arterial tumors is likely underestimated because of the misdiagnosis of thromboembolic disease. Findings such as fever, weight loss, and anemia may help distinguish pulmonary arterial malignancy from obstruction due to congenital or thromboembolic disease [1]. Echocardiography may allow visualization of the tumor within the right ventricular outflow tract. Advances in cardiac MRI imaging have improved the rate of preoperative diagnosis substantially in recent years. Factors that favor diagnosis of pulmonary artery sarcoma are heterogeneous soft tissue density, vascular distension, and enhancement of the gadolinium contrast [1]. Although pulmonary artery sarcoma carries a poor prognosis because of difficulty in diagnosis and high metastatic potential, surgical resection offers the best chance of survival. In a review of 93 cases of pulmonary artery sarcoma, Kruger and associates [4] reported a median survival of 1.5 months after diagnosis. However, the 27 patients who underwent surgical resection at the time of diagnosis survived an average of 10 months. The role of chemotherapy in the treatment of pulmonary artery sarcoma is unclear. A recent review reported about a 50% response rate to palliative chemotherapy with anthracyclines and ifosfamide in patients with advanced pulmonary artery sarcoma [5]. There is little information to direct treatment or predict outcome in this patient, given the rarity of this tumor in
FEATURE ARTICLES
Ann Thorac Surg 2008;85:1092– 4
1094
CASE REPORT KADOHAMA ET AL VAC FOR PEDIATRIC MEDIASTINITIS
children. His tumor burden has steadily decreased with surgery and chemotherapy. Given the experience in adults, his long-term prognosis is poor. It is likely that his original pulmonic stenosis at 2 months of age was due to tumor growth at the valve. Therefore, pulmonary artery sarcoma should be considered in the differential diagnosis of pulmonary stenosis presenting in infancy.
References
FEATURE ARTICLES
1. Yi ES. Tumors of the pulmonary vasculature. Cardiol Clin 2004;22:431– 40. 2. Farooki Z, Chang C, Jackson WL, et al. Primary pulmonary artery sarcoma in two children. Pediatr Cardiol 1988;9:243–51. 3. Nonomura A, Kurumaya H, Kono N, et al. Report of two autopsy cases studied by immunohistochemistry and electron microscopy and review of 110 cases reported in the literature. Acta Pathol Jpn 1988;38:883–96. 4. Kruger I, Borowski A, Horst M, de Vivie ER, Theissen P, Gross-Fengels W. Symptoms, diagnosis, and therapy of pulmonary sarcomas of the pulmonary artery. Thorac Cardiovasc Surg 1990;38:91–5. 5. Manso L, Alvarez E, Quintela M, Cortes-Funes H, Hitt R. Primary pulmonary artery sarcoma: report of three cases and review of the literature. Clin Lung Cancer 2007;8:277– 81.
Vacuum-Assisted Closure for Pediatric Post-Sternotomy Mediastinitis: Are Low Negative Pressures Sufficient? Takayuki Kadohama, MD, PhD, Nobuyuki Akasaka, MD, PhD, Akira Nagamine, MD, Keisuke Nakanishi, MD, Keiko Kiyokawa, MD, Kazutomo Goh, MD, PhD, and Tadahiro Sasajima, MD, PhD Departments of Surgery and Emergency Medicine, Asahikawa Medical University, Asahikawa, Japan
We present 3 cases of pediatric post-sternotomy mediastinitis treated by a vacuum-assisted closure (VAC). The patients 2 girls, aged 6 months and 10 months, and a 2-year-old boy. The onset of infection was at 9, 14, and 32 postoperative days. The culture examination detected coagulase-negative Staphylococci strains in 2 cases, and Staphylococcus aureus in 1 case. A VAC was performed at ⴚ50 mm Hg for 10, 12, and 7 days. The wounds were closed without vascularized soft tissue. A VAC under a low negative pressure is a useful and safe procedure for the management of pediatric post-sternotomy mediastinitis. (Ann Thorac Surg 2008;85:1094 – 6) © 2008 by The Society of Thoracic Surgeons Accepted for publication Sept 4, 2007. Address correspondence to Dr Kadohama, Asahikawa Medical University, Department of Surgery, Midorigaoka-Higashi 2-1-1-1, Asahikawa, 078-8510, Japan; e-mail: [email protected].
© 2008 by The Society of Thoracic Surgeons Published by Elsevier Inc
Ann Thorac Surg 2008;85:1094 – 6
P
ost-sternotomy mediastinitis, also commonly called deep sternal wound infection, is one of the most feared complications in patients undergoing cardiac surgery [1]. At the present time, there is no general consensus regarding the appropriate surgical approach to mediastinitis after cardiac surgery. Vacuum-assisted closure (VAC) is a novel treatment with an ingenious mechanism, and recent publications have demonstrated encouraging clinical results [2]. However, the validity of VAC therapy for managing pediatric post-sternotomy mediastinitis remains to be confirmed because there have been very few reports published on this topic [3, 4]. We herein describe 3 cases of pediatric post-sternotomy mediastinitis treated by VAC therapy. Patient 1 was a 6-month-old girl who underwent a bilateral bidirectional Glenn shunt procedure for a tricuspid atresia with a concomitant pulmonary atresia. The surgery was performed urgently owing to clinical deterioration, as evidenced by the progression of cyanosis, which required intubation and mechanical control of ventilation. Nine days after surgery, she was noted to have a purulent discharge from the median sternal wound and a high-grade fever. Removal of the sternal wires and surgical debridement were performed. Thereafter, polyurethane foam, which was shaped to fit the defect, was placed within the cavity. The area was covered with an adhesive drape, and suction drainage was carried out at ⫺50 mm Hg for 10 days in addition to the administration of appropriate antibiotics. Coagulasenegative Staphylococci strains were detected by the culture examination. The polyurethane foam was replaced every few days. The VAC therapy lasted for 10 days. The wound was closed without any surgical revisions at 3 days after the discontinuation of VAC (Fig 1). Patient 2 was a 2-year old boy who underwent a pacemaker implantation using myocardial leads for a sick sinus syndrome. A total anomalous pulmonary venous connection repair and a release of a pulmonary venous obstruction were performed twice before this operation. The patient was noted to have a high-grade fever and a purulent discharge from the median sternal wound 14 days after surgery. Coagulase-negative Staphylococci strains were detected by the culture examination. Debridement of infected tissue was performed at the time of VAC application. We employed VAC therapy for 12 days until the wound became smaller and granulation tissue proliferated, in addition to the administration of appropriate antibiotics. However, there was still a small defect in the epithelialization. Finally, a second primary closure was performed at 4 weeks after VAC application. Patient 3 was a 10-month old girl who underwent a ventricular septal defect (VSD) repair. After she was discharged from the hospital (postoperative day 32), she was noted to have a high-grade fever and a purulent discharge from the median sternal wound. Staphylococcus aureus was detected by the culture examination. Debridement of the infected tissue was performed at the time of 0003-4975/08/$34.00 doi:10.1016/j.athoracsur.2007.09.004