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• W2953209432 abstract "Surgical correction of Cantrell syndrome is often associated with an extremely high mortality rate due to the possibility of wound infection or the severity of cardiac anomalies. We report a case of Norwood operation and repositioning of the heart successfully performed 1 day after the birth of a neonate with pentalogy of Cantrell. The patient had double-outlet right ventricle, subaortic stenosis, aortic valve stenosis, hypoplastic aortic arch, and coarctation of the aorta. The patient underwent the Glenn operation at the age of 1 year and is now waiting for the Fontan operation. Surgical correction of Cantrell syndrome is often associated with an extremely high mortality rate due to the possibility of wound infection or the severity of cardiac anomalies. We report a case of Norwood operation and repositioning of the heart successfully performed 1 day after the birth of a neonate with pentalogy of Cantrell. The patient had double-outlet right ventricle, subaortic stenosis, aortic valve stenosis, hypoplastic aortic arch, and coarctation of the aorta. The patient underwent the Glenn operation at the age of 1 year and is now waiting for the Fontan operation. Cantrell syndrome is a rare congenital syndrome that was first reported by Cantrell and colleagues in 1958.1Cantrell J.R. Haller J.A. Ravitch M.M. A syndrome of congenital defects involving the abdominal wall, sternum, diaphragm, pericardium, and heart.Surg Gynecol Obstet. 1958; 107: 602-614PubMed Google Scholar They defined the syndrome as a pentalogy composed of defects of the abdominal wall, diaphragm, pericardium, and lower sternum and congenital heart disease. Its prognosis is poor and depends on the severity of the cardiac anomaly and defective skin area in terms of the susceptibility to infection.2Vazquez-Jimenez J.F. Muehler E.G. Daebritz S. et al.Cantrell’s syndrome: a challenge to the surgeon.Ann Thorac Surg. 1998; 65: 1178-1185Abstract Full Text Full Text PDF PubMed Scopus (82) Google Scholar, 3O'Gorman C.S. Tortoriello T.A. McMahon C.J. Outcome of children with pentalogy of Cantrell following cardiac surgery.Pediatr Cardiol. 2009; 30: 426-430Crossref PubMed Scopus (40) Google Scholar, 4van Hoorn J.H. Moonen R.M. Huysentruyt C.J. van Heurn L.W. Offermans J.P. Mulder A.L. Pentalogy of Cantrell: two patients and a review to determine prognostic factors for optimal approach.Eur J Pediatr. 2008; 167: 29-35Crossref PubMed Scopus (88) Google Scholar Herein, we report a case wherein Norwood operation and repositioning of the heart into the thorax were successfully performed 1 day after the birth of a neonate with pentalogy of Cantrell. A 1-day-old boy was delivered through a scheduled cesarean section owing to fetal diagnosis of pentalogy of Cantrell, double-outlet right ventricle, subaortic stenosis, aortic valve stenosis, hypoplastic aortic arch, and coarctation of the aorta. Because of the defects of the lower portion of the sternum, the heart was protruding and covered by an extremely thin skin. The supraumbilical abdominal part was covered by the amniotic membrane. The patient’s body weight was 2.616 g. Based on computed tomography, the sizes of the ascending aorta, transverse aorta, and isthmus were found to be 4.3 mm, 2.4 mm, and 2.0 mm, respectively (Figure 1). Echocardiography revealed that the flow in the left common carotid artery was supplied from the patent ductus arteriosus, and the right ventricle was hypoplastic. The pulmonary artery flow showed an increase immediately after birth; therefore, nitrogen inhalation was administered to control the high flow status, without avail. An urgent Norwood operation and repositioning of the heart into the thorax were performed. The skin incision line was median in the upper portion of the sternum and right oblique along the right costal arch in the lower portion (Figures 2A, 2B). We dissected the ectopia cordis from the thin skin, incised the left side of the pericardium, and repositioned the heart in the left thorax (Figures 2C, 2D). During the procedure, we paid careful attention to the lack of remarkable changes in the intracardiac flow pattern on transesophageal echocardiography. Sano shunting (right ventricle to pulmonary artery shunt) was conducted. The operation, cardiopulmonary bypass, and cardiac arrest lasted for 341, 208, and 74 minutes, respectively. The chest was closed after 5 days postoperatively. The patient was extubated 90 days after the surgery because of tracheomalacia and hydrocephalus, which were not related to the operation. He underwent Glenn operation at the age of 1 year and is now waiting for the Fontan operation. This report describes the Norwood operation successfully performed for Cantrell syndrome. Although bilateral pulmonary artery banding might have been another option as a staged operation, we considered that multiple surgical interventions should be avoided owing to the high risk of wound infection developing. Furthermore, there was a possibility of critical hemodynamic changes by stenosis or regurgitation of the valves due to repositioning of the heart; transesophageal echocardiography was useful for constant monitoring. The procedure must be performed to protect the heart from future infections and trauma if the hemodynamic change is acceptable. Another problem would be the defective skin area in the lower part of the sternum; a larger defective area indicates a higher risk of infection and larger adhesion area between the skin and heart. Ito and colleagues5Ito H. Ota N. Murata M. Sakamoto K. Fontan operation for the Cantrell syndrome using a clamshell incision.Interact Cardiovasc Thorac Surg. 2013; 17: 754-756Crossref PubMed Scopus (2) Google Scholar described the clamshell incision by which they could avoid a zigzag incision and perform bilateral cavopulmonary anastomosis. We did not use the clamshell incision to preserve pulmonary function and to minimize wound infection by the large incision line. Instead, we adopted a midline incision in the upper part of the sternum and a right oblique incision along the right costal arch in the lower portion. Even with some difficulties in dissection between the skin and heart, we could preserve the skin in good condition; therefore, we could close the incision without using any artificial materials. The authors wish to thank Enago for their English language editing services." @default.
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• W2953209432 date "2020-02-01" @default.
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• W2953209432 title "Norwood Operation of a Neonate With Pentalogy of Cantrell" @default.
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