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Asian Cardiovasc Thorac Ann 2001;9:232-233
© 2001 Asia Publishing EXchange Pte Ltd

CASE STUDY

Repair of Double Coaraction or Aorta With Associated Anomalies

Harinder Singh Bedi, MCh, Maninder Singh Kalkat, MCh, Ved Prakash Sharma, DM, Vijay Mahajan, MD

Tagore Heart Care & Research Centre Jalandhar, Punjab, India
For reprint information contact: Harinder Singh Bedi, MCh Tel: 91 172 39 6235 Fax: 91 172 39 6221 email: bedi{at}fortis.co.in Department of Cardiovascular Surgery, Fortis Heart Institute, Sector 62, Phase VIII, Mohali, Punjab 160055, India.

   Abstract
TOP
 Abstract
INTRODUCTION
 CASE REPORT
 DISCUSSION
 References
 
An 8-year-old boy who had coarctation of the aorta with two stenotic ridges, a stenotic left subclavian artery, and absence of collaterals, underwent successful surgical repair. Both ridges showed histopathological features of true coarctation.


   INTRODUCTION
TOP
 Abstract
 INTRODUCTION
CASE REPORT
 DISCUSSION
 References
 
Coarctation of the aorta with associated anomalies comprising 2 stenotic ridges, a stenotic left subclavian artery, and absence of collaterals, presented some unique surgical problems.


   CASE REPORT
TOP
 Abstract
 INTRODUCTION
 CASE REPORT
DISCUSSION
 References
 
An 8-year-old boy with symptoms of headaches and easy fatigability was referred because of coarctation of the aorta. On examination, the left brachial pulse was not palpable. Echocardiography indicated 2 ridges in the thoracic aorta (Figure 1Go). Angiography revealed severe coarctation (gradient of 110 mm Hg) with the left subclavian artery arising below the segment of coarctation. No collaterals were seen on either side on chest radiography or angiography. The absence of collaterals on the right side could not be explained. The special surgical problems in this case were the risk of paraplegia due to the absence of collaterals, the presence of 2 ridges to be dealt with, and the need to treat the left subclavian artery stenosis. The findings were confirmed at surgery via a left posterolateral thoracotomy. Severe poststenotic dilatation, stenotic origin of the left subclavian artery from the distal segment, and separate origin of the left vertebral artery were noted (Figure 22Go). To reduce the risk of paraplegia, the patient was cooled by reducing the ambient temperature, using a cold water blanket, and applying topical ice slush in the pleural cavity. In addition, an indigenously assembled shunt was inserted after heparinization, to divert blood from the proximal to the distal segment during crossclamping. The shunt com-prised 2 left ventricular vents connected together. After heparinization (1 mg•kg–1), insertion of the shunt, and clamping, the segment of coarctation was excised as a ring (Figure 3Go). The left subclavian artery with stenosis at its origin was detached along with the coarctation segment. From within the proximal segment, the 2nd ridge (which was well above the coarctation site) was easily and completely excised. End-to-end repair was performed with interrupted sutures anteriorly. The subclavian artery was then reimplanted in the proximal segment. The patient had bounding left radial artery and lower limb pulsations at the end of the procedure and made an uneventful recovery. At the 1-year follow-up, he was off all antihypertensive medications. Histopathology showed an infolding of the aortic media with a ridge of intimal hyperplasia in both segments.



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  		Figure 1. Preoperative echocardiogram showing the 2 ridges.

 


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  		Figure 2. Intraoperative photograph showing the poststenotic dilated segment (DS), the left subclavian artery (SA), separate origin of the left vertebral artery (VA), and the ligamentum arteriosum (LA).

 


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  		Figure 3. Excised specimen of the ring of coarctation (C) with stenotic origin of the left subclavian artery (DS) and part of the excised proximal ridge (arrow).

 

   DISCUSSION
TOP
 Abstract
 INTRODUCTION
 CASE REPORT
 DISCUSSION
References
 
The embryological basis of coarctation is still unknown.1–4 Developmentally, several structures are involved in the region where a coarctation develops. The distal part of the 6th aortic arch (ductus arteriosus) unites with the dorsal aorta just distal to the entrance of the 4th aortic arch, and the left subclavian artery arises from the dorsal aorta in this area. The causative factors involved in the formation of the adult form of coarctation are still debatable. According to Ho and Anderson,4 it is due to the same process as that is responsible for obliteration of the ductus arteriosus; this implies extension of the tissue of the ductus into the aorta. The ridge or shelf is believed to represent the original wall of the distal left 6th aortic arch. It is thought to be due to an error in development at the junction of the 4th and 6th aortic arches; after birth, traction of the obliterating ductus causes kinking and narrowing of the descending limb of the aortic arch.1 Alternatively, a coarctation may be due to the absence, atrophy, and imperfect development of the 4th left aortic arch.1

In view of the lack of consensus on the embryological basis for adult coarctation, the development of a double coarctation in this case is even more difficult to explain. Double coarctations have been reported in autopsies, and coarctation proximal to the left subclavian artery, although rare, is known to occur (in 1% of all cases). However, we believe this is the first case of a double ridge with stenosis of the left subclavian artery to have been successfully treated surgically.3,5,6


   References
TOP
 Abstract
 INTRODUCTION
 CASE REPORT
 DISCUSSION
 References
 

  1. Hudson RE. Cardiovascular pathology. London: Edward Arnold, 1965:2050–68.

  2. Perloff JK. The clinical recognition of congenital heart disease. 4th ed. Philadelphia: Saunders, 1994:132–69.

  3. Kirklin JW, Barratt-Boyes BG. Cardiac surgery. 2nd ed. New York: Churchill-Livingstone, 1993:1263–325.

  4. Ho SY, Anderson RH. Coarctation, tubular hypoplasia and the ductus arteriosus. Histologic study of 35 specimens. Br Heart J 1979;41:268–74.[Abstract/Free Full Text]

  5. Braunwald E. Heart disease. 5th ed. Philadelphia: Saunders, 1997:911–3.

  6. Stark J, de Leval M. Surgery for congenital heart defects. 2nd ed. Philadelphia: Saunders, 1994:285–98.





This Article
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Maninder Singh Kalkat
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Right arrow Articles by Mahajan, V.
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Right arrow Articles by Bedi, H. S.
Right arrow Articles by Mahajan, V.
Related Collections
Right arrow Congenital - acyanotic

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