Ann Thorac Surg 2003;75:1308-1310
© 2003 The Society of Thoracic Surgeons
Case report
Lobar emphysema due to ductus arteriosus compressing right upper bronchus in an infant with congenital heart disease
Takashi Hishitani, MD*a,
Kiyoshi Ogawa, MDa,
Kenji Hoshino, MDa,
Yuzuru Nakamura, MDb,
Tadashi Iwanaka, MDc,
Kayo Masago, MDd,
Misako Suzuki, MDd
a Division of Cardiology, Saitama, Japan
b Division of Cardiovascular Surgery, Saitama, Japan
c Division of Surgery, Saitama, Japan
d Division of Anesthesiology, Saitama Children’s Medical Center, Iwatsuki, Saitama, Japan
Accepted for publication October 8, 2002.
* Address reprint requests to Dr Hishitani, Division of Cardiology, Saitama Children’s Medical Center, Magome 2100, Iwatsuki, Saitama 339-8551, Japan
e-mail: a1075097{at}pref.saitama.jp
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Abstract
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A 1-month-old boy with tetralogy of Fallot, pulmonary atresia, right aortic arch, and right ductus arteriosus, exhibited progressive right upper lobar emphysema since his birth. The emphysema was caused by the right ductus arteriosus compressing the right upper bronchus. After division of the ductus arteriosus the emphysema completely regressed. We should explore the cause of lobar emphysema thoroughly before lobectomy especially when it is extrinsic. The emphysema may regress by eliminating the extrinsic factor.
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Introduction
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Congenital lobar emphysema is a rare clinical condition that often requires lobectomy in early infancy. Bronchial cartilaginous dysplasia is often associated with this condition, but in half the affected infants etiology remains undetermined [1]. An extrinsic obstruction, such as dilated pulmonary arteries or left atrium associated with ventricular septal defect, a patent ductus arteriosus, or tetralogy of Fallot with dilated pulmonary arteries may cause this condition [1–3]. We report here an infant with right upper lobar emphysema caused by a dilated ductus arteriosus compressing the right upper bronchus.
A 1-month-old boy with tetralogy of Fallot, pulmonary atresia, right aortic arch, and right ductus arteriosus had progressive right upper lobar emphysema since his birth (Fig 1).
He didn’t exhibit any apparent symptoms except mild cyanosis due to congenital cardiac defect. The right upper bronchus could not be recognized in chest computed tomographic scan (CT), and we speculated that the dilated right ductus arteriosus, which was located just on the right bronchus, was compressing the right upper bronchus (Fig 2).
Angiography of the descending aorta revealed a long dilated ductus arteriosus curving toward the right upper bronchus (Fig 3).
After the plasty of the right pulmonary artery, the division of the long dilated ductus arteriosus that bent onto the right bronchus, and the creation of the aortopulmonary anastomoses under cardiopulmonary bypass, the patient returned to ICU without any respiratory trouble. Postoperative bronchofiberscopy illustrated no abnormality in the right upper bronchus. The emphysema gradually disappeared in a few days, and the patient was discharged without any symptoms.
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Fig 1. Chest roentgenogram at 1 month of age, illustrating right upper lobar emphysema and downward shift of hair line (white arrow).
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Fig 2. Chest computed tomographic scan revealing long dilated ductus arteriosus originating from descending aorta, passing by right bronchus, and connecting to right pulmonary artery. (DA = ductus arteriosus; DAo = descending aorta; LB = left bronchus; LPA = left pulmonary artery; RB = right bronchus; RPA = right pulmonary artery.)
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Fig 3. Angiography of descending aorta illustrating long dilated ductus (arrow) curving toward right upper bronchus. (DA = ductus arteriosus; LPA = left pulmonary artery; RPA = right pulmonary artery.)
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Comment
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Congenital heart disease is associated with 12% to 20% of patients with lobar emphysema [1, 3], usually resulting from hypertensive or dilated pulmonary arteries associated with ventricular septal defect or patent ductus arteriosus [1, 2, 5]. Several cases of lobar emphysema with tetralogy of Fallot have been reported [2–5], and most of them were combined with dilated pulmonary arteries. There were four cases of tetralogy of Fallot without any description of dilated pulmonary arteries. In two of them, right aortic arch and right upper lobar emphysema were noted. One of them underwent urgent right upper lobectomy at the age of 1 month, the other had no lobectomy and had persistent radiolucency of the right upper lung, which was not clinically significant. There was no information about whether these patients had ductus arteriosus, and the causes of the lobar emphysema were unknown [3, 5]. There have been no reports claiming that a dilated ductus arteriosus caused lobar emphysema by compressing one of the branches of the bronchus. In the present patient emphysema was found in the right upper lobe, which is rare site in cases of lobar emphysema with congenital heart disease [2]. The most frequent site for emphysema is the right middle lobe, where the right middle bronchus is often compressed by dilated pulmonary vessels [2]. The present patient had no dilated pulmonary vessels but had a dilated right ductus arteriosus, which is located near the right upper bronchus. If emphysema is found in the right upper lobe in congenital heart disease patients with right aortic arch, and there is no sign of vascular ring, we should take it into account that right ductus arteriosus might compress right upper lobe.
Careful anesthesia management is necessary during the operation for lobar emphysema. Nitrous oxide should not be used, and spontaneous respiration with minimum assisted ventilation may be preferred before thoracotomy in severe cases [6]. In the present patient, preparing emergency thoracotomy and cardiopulmonary bypass, we cautiously performed the induction of anesthesia with fentanyl, added pancuronium bromide, and kept gentle ventilation with monitoring the I/E ratio under tracheal intubation. Anesthetic management was successful and uneventful.
Postoperative bronchofiberscopy revealed no anatomic abnormalities inside the right bronchus, and the emphysema regressed completely in a few days after the division of ductus arteriosus. Urgent lobectomy may be necessary for the infant with severe respiratory distress, but we should be careful in performing lobectomy because lobar emphysema from extrinsic cause can regress without lobectomy after the cause is removed.
Conclusion
We presented a case of lobar emphysema due to the right ductus arteriosus compressing the right upper bronchus in a infant with congenital heart disease. We should explore the cause of lobar emphysema thoroughly before lobectomy especially when it is extrinsic. The emphysema may regress by eliminating the extrinsic factor.
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References
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- Hendren W.H., Mckee D.M. Lobar emphysema in infancy. J Pediatr Surg 1966;1:24-38.
- Stanger P., Lucas R.V., Edwards J.E. Anatomic factors causing respiratory distress in cyanotic congenital cardiac disease. Pediatr 1969;43:760-769.[Abstract/Free Full Text]
- Roguin N., Peleg H., Lemer J., Naveh Y., Riss E. The value of cardiac catheterization and cineangiography in infantile lobar emphysema. Pediatr Radiol 1980;10:71-74.[Medline]
- Jones J.C., Almond C.H., Snyder H.M., Meyer B.W., Patrick J.R. Lobar emphysema and congenital heart disease in infancy. J Thorac Cardiovasc Surg 1965;49:1-10.
- Pierce W.S., Paredes C.G., Friedman S., Waldhausen J.A. Concomitant congenital heart disease and lobar emphysema in infants. Ann Surg 1970;172:951-956.[Medline]
- Al-Salem A.H., Adu-Gyamfi Y., Grant C.S. Congenital lobar emphysema. Can J Anaesth 1990;37:377-379.[Abstract/Free Full Text]
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Abstract
Introduction
