Interact CardioVasc Thorac Surg 2009;9:366-367. doi:10.1510/icvts.2009.202234
© 2009 European Association of Cardio-Thoracic Surgery
Case report - Vascular thoracic |
Acute aortic dissection occurring during the butterfly stroke in a 12-year-old boy
Keiji Uchida*,
Kiyotaka Imoto,
Hiromasa Yanagi and
Koichiro Date
Yokohama City University Medical Center, Cardiovascular Center, 4-57 Urafune-cho, Minami-ku, Yokohama, 232-0024, Japan
Received 8 January 2009;
received in revised form 30 April 2009;
accepted 1 May 2009
*Corresponding author. Tel.: +81-45-261-5656; fax: +81-45-261-9162.
E-mail address: uchida{at}urahp.yokohama-cu.ac.jp(K. Uchida).
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Abstract
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A 12-year-old boy had severe chest and back pain of sudden onset while practicing the butterfly stroke in a swimming class. Computed tomography revealed an intimal flap in the descending thoracic aorta with massive right hemothorax. A ruptured type B acute aortic dissection was diagnosed, and then he collapsed. We totally replaced the descending aorta with a woven polyester prosthetic graft during deep hypothermic circulatory arrest. Hemostasis was achieved, but consciousness was not regained after operation, and multiple organ failure occurred. He died on the fifth postoperative day. He and his family had no history of cardiovascular disease. It seems that the swimming provoked a severe Valsalva maneuver, raising blood pressure acutely and thereby leading to dissection. This is then analogous to the propensity for dissection during intense isometic exercise such as weightlifting.
Key Words: Acute aortic dissection in children; Butterfly stroke; Swimming
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1. Introduction
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Acute aortic dissection in childhood is a rare condition that can be associated with many risk factors, including congenital cardiovascular disorders, connective tissue disorders, trauma, and hypertension. We describe our experience with a 12-year-old boy in whom acute aortic dissection developed while he was practicing the butterfly stroke. The patient had no other known risk factors.
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2. Clinical case
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The patient was a 12-year-old boy who had a history of Epstein–Barr viral infection and surgery for cryptorchidism. He and his family had no history of cardiovascular disease. While practicing the butterfly stroke in a swimming class at his junior high school, he suddenly experienced severe chest and back pain. He first consulted an orthopedist, but no abnormalities were found. He then went to a pediatrician. His height was 150 cm, and the body weight was 50 kg. His physique was muscular and well developed for his age. His consciousness was clear, and systolic blood pressure was 160 mmHg. The patient complained of abdominal pain and was given an enema, with no improvement in symptoms. Finally, enhanced computed tomography revealed an intimal flap in the descending thoracic aorta. Acute aortic dissection was diagnosed. The pediatrician could not diagnose the type of dissection because of motion artifacts caused by the ascending aorta. The left kidney was not enhanced by contrast medium, indicating that the dissection involved the left renal artery. After computed tomographic scanning, the blood pressure dropped to 80 mmHg, and the patient became drowsy. He was emergently transferred to our hospital for further evaluation and treatment.
On admission, a chest X-ray film showed massive right hemothorax. Computed tomography was therefore repeated. The aortic dissection was found to be Stanford type B and ruptured into the right pleural cavity (Fig. 1). On entering the operating room, the patient collapsed. Standard cardiopulmonary resuscitation was followed by an emergent left thoracotomy, and extracorporeal circulation was started. Transesophageal echocardiography showed that the entry was located just below the left subclavian artery, and the true lumen of the descending aorta was severely narrowed. We found a tear of the adventitia in the lower part of the descending aorta. We therefore totally replaced the descending aorta with a woven polyester prosthetic graft during deep hypothermic circulatory arrest. Because the diameter of the true lumen of the descending aorta was 11 mm, we used a graft 12 mm in diameter. The patient was weaned from cardiopulmonary bypass, and hemostasis was achieved. However, consciousness was not regained after operation, and multiple organ failure occurred. He died on the fifth postoperative day. Histopathological examination of a specimen of the dissected aortic wall obtained during surgery revealed no signs of medial degeneration or necrosis (Fig. 2).
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Fig. 1. A computed tomographic scan, showing a Stanford type B aortic dissection, rupturing into the right pleural cavity.
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Fig. 2. Histopathological findings of the flap (hematoxylin-eosin stain), showing no signs of medial degeneration or necrosis.
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3. Discussion
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Aortic dissection is rare in children. Among a total of 1085 cases of dissecting aortic aneurysm, 38 (3.5%) occurred in persons 19 years of age or younger [1]. During a 1-year period, two of 5658 deaths in New York State among persons aged 0–19 years were caused by aortic dissection [1]. Prompt diagnosis may be life-saving because untreated aortic dissection has extremely poor outcomes [1]. Fikar et al. reported that most children with aortic dissection have known predisposing risk factors, such as cardiovascular anomalies, Marfan's syndrome or other connective tissue disorders, hypertension, trauma, and weight training [1]. Zalzstein et al. reported 13 patients younger than 25 years who had acute or chronic aortic dissections between 1970 and 2000. Congenital cardiovascular anomalies were diagnosed in five of these patients, Marfan's syndrome in four, and blunt trauma to the chest in three. No risk factors were identified in the remaining patient, a 17-year-old girl [2].
Our patient had been in good health, with no congenital abnormality. Histopathological examination of the aortic wall showed no evidence of medial necrosis or degeneration. Acute aortic dissection occurred in him during the training of the butterfly stroke.
Many reports point out the relationship between aortic dissection and weightlifting [3]. In our patient, it seems that the swimming provoked a severe Valsalva maneuver, raising blood pressure acutely and thereby leading to dissection. This is then analogous to the propensity for dissection during intense isometic exercise such as weightlifting. From the viewpoint of sports medicine, the butterfly stroke might cause shoulder pain and spondylolysis [4], but has not been associated with aortic dissection previously.
The patient's muscular physique was adult-like, but the diameter of the true lumen of the descending thoracic aorta was only 11 mm. His underdeveloped thoracic aorta might have been relatively frail and susceptible to acute blood pressure rising. Swimming coaches and pediatricians should recognize that hard swimming training like the butterfly stroke is a very rare risk factor for aortic dissection in children.
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References
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- Fikar CR, Koch S. Etiologic factors of acute aortic dissection in children and young adults. Clin Pediatr (Phila) 2000;39:71–80.[Abstract/Free Full Text]
- Zalzstein E, Hamilton R, Zucker N. Aortic dissection in children and young adults: diagnosis, patients at risk, and outcomes. Cardiol Young 2003;13:341–344.[Medline]
- Hatzaras I, Tranquilli M, Coady M. Weight lifting and aortic dissection: more evidence for connection. Cardiology 2007;107:103–106.[CrossRef][Medline]
- Johnson JE, Sim FH, Scott SG. Musculoskeletal injuries in competitive swimmers. Mayo Clin Proc 1987;62:289–304.[Medline]
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Abstract
1. Introduction