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Acquired aortic coarctation: A rare case of supraortic valve stenosis
International Journal of Cardiology 156 (2012) e62–e64
Contents lists available at SciVerse ScienceDirect
International Journal of Cardiology journal homepage: www.elsevier.com/locate/ijcard
Letter to the Editor
Acquired aortic coarctation: A rare case of supraortic valve stenosis L. Di Marco a,⁎, D. Pacini a, R. Lorusso b, G.F. Pasini c, G. Zanini c, A. Pantaleo a, R. Di Bartolomeo a a b c
Cardiac Surgery Department, S. Orsola-Malpighi Hospital, University of Bologna, Bologna, Italy Cardiac Surgery Unit, Civic Hospital, Brescia, Italy Cardiology Unit, “La Memoria” Hospital, Gavardo, Italy
a r t i c l e
i n f o
Article history: Received 2 August 2011 Accepted 20 August 2011 Available online 13 September 2011 Keywords: Supravalvular stenosis Aortic valve Teflon felt Ascending aorta
Acquired supravalvular aortic stenosis (SVAS) is a very rare complication following cardiac surgery. Usually, SVAS is a congenital condition characterized by narrowing of the aorta close to its origin. It is either isolated or part of Williams syndrome. Moreover, isolated iatrogenic or acquired case of SVAS has been also reported. After an accurate review of the literature, this is one of the few cases of symptomatic supravalvular aortic stenosis after replacement of the ascending aorta. A 65-year-old Asian female was referred to our institution from other hospital with a diagnosis of supravalvular aortic stenosis. As cardiovascular risk factors she was hypertensive and diabetic. In 2010, we operated the patient for elective replacement of supracommissural ascending aorta, aortic arch and proximal portion of the descending thoracic aorta with frozen elephant trunk technique using a 28 mm E-vita Open Plus (Jotec GmbH, Germany) because of chronic aneurysm of the thoracic aorta. Our technique for complete replacement of the thoracic aorta with frozen elephant trunk has been already described [1].Once the graft was delivered in the proximal portion of the descending thoracic aorta and the aortic arch replaced, the Dacron graft was sutured to the previously prepared supracommissural ascending aorta with a double internal–external Teflon felt used to reinforce the suture. Postoperatively, the patient had an uneventful recovery, the angio-Computed Tomography (CT) scan showed a slight reduction in size of the ascending aorta lumen at the proximal site of the anastomosis and a complete exclusion of the aneurysmal dilatation distally to
⁎ Corresponding author at: Cardiac Surgery Department, University of Bologna, S. Orsola-Malpighi Hospital, Via Massarenti, 9, 40138 Bologna, Italy. Tel.: +39 0516363361; fax: +39 051345990. E-mail address: [email protected] (L. Di Marco). 0167-5273/$ – see front matter © 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2011.08.069
the E-vita prosthesis so she was discharged (Fig. 1). One year later, she was hospitalized for the onset of dyspnea on exertion and for occasional episodes of dizziness. Transthoracic echocardiography (TTE) showed left ventricular normal function (ejection fraction 56%), the aortic valve showed a mild aortic regurgitation and the tricuspid valve a moderate regurgitation with severe pulmonary hypertension (peak value 80 mm Hg).Doppler revealed a turbulent flow in the proximal portion of the ascending aorta with a pressure gradient between left ventricle and the aorta of 109 mm Hg. The patient underwent to transesophageal echocardiography (TEE) which revealed an ascending aorta acceleration flow at 3.7 cm from the aortic annulus with a persistence of transaortic supravalvular gradient of 93 mm Hg. The angio-CT scan control revealed a significative stenosis of the ascending aorta at the proximal suture with Dacron portion of the E-vita prosthesis at 3 cm from the aortic annulus with a severe reduction of the lumen at 1.1 cm. It also showed a type 1 endoleak distally to the E-vita graft with an aneurysm of the distal portion of the descending thoracic aorta of 5.9 cm. In the light of this instrumental finding, the patient was then referred at our Institution with a diagnosis of supravalvular aortic stenosis. On admission, the patient was asymptomatic. An electrocardiogram showed left ventricular hypertrophy.TTE confirmed severe iatrogenic supravalvular stenosis attributable to surgical material at 3 cm from the valvular plane with a peak gradient pressure of 90 mm Hg (Fig. 2A). The patient repeated the angio-CT scan which confirmed a severe caliber-shrinkage of the ascending aorta at proximal anastomosis site (minimum diameter: 11–12 mm) and an increase of the aneurismal dilatation of the descending thoracic aorta distally to the E-vita prosthesis (Fig. 2B–C). The patient underwent reoperation. The proximal anastomosis was opened, and we detected that the inner teflon felt created a supravalvular labrum causing the supraortic valve stenosis. It was then removed. The proximal supracoronary aorta was suitably shaped in oblique fashion in order to make wider anastomosis. The proximal anastomosis between the graft and the ascending aorta was then completed and reinforced with the only external Teflon felt. Following an uneventful recovery period, the patient underwent to TTE control which showed the absence of the gradient at the site of proximal anastomosis. The patient was then discharged. Postoperative bleeding represents one of the main problem of aortic surgery. To reduce its incidence, various techniques have been developed. The use of external or double Teflon felt, internal and external, at the site of anastomosis, represents one of these. A double Teflon felt strip may be used outside and inside the aorta, and fixed with a horizontal mattress suture. Care must be taken when the
L. Di Marco et al. / International Journal of Cardiology 156 (2012) e62–e64
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Fig. 1. Angio CT scan; A–B): The CT scan shows a slight size-reduction at the proximal site of the anastomosis (diameter: 23 mm) between native ascending aorta and Dacron prosthesis. Distally to the E-vita prosthesis, complete exclusion of the aneurismal dilatation of the descending thoracic aorta.
suture is tied in order to avoid the excessive narrowing of the aorta: in fact, if the mattress suture is too much pulled, the lumen of the aorta may be narrowed, especially if the native aorta is small. The present report shows a very unusual complication following ascending aorta replacement, presenting as supravalvular aortic stenosis and demonstrated by echocardiographic finding of a flow acceleration with a gradient pressure at the level of the proximal anastomosis between supracommissural portion of the ascending aorta and proximal Dacron portion of the E-vita prosthesis. Usually, SVAS is a congenital condition and it is strongly associated with Williams syndrome, a defect involving the elastin gene, although isolated iatrogenic or acquired case of SVAS have been previously reported [2,3]. SVAS may result from the kinking caused by redundant donor ascending aorta tissue following heart transplantation or from peri-aortic pseudoaneurysm compressing the aortic graft [3,4].The present report describes a case of iatrogenic stenosis of the supracoronary ascending aorta after replacement for chronic aneurysm, due to the healing process at the level of the proximal anastomosis which caused a stiffening of the inner felt with consequent severe reduction of the ascending aorta lumen at the sinotubular junction. Previous authors described supraaortic stenosis originating from double Teflon felt associated with biological glue used to reinforce the proximal anastomosis in a case of type A aortic dissection [5]. As they pointed out, the Teflon felt used to reinforce the dissection following glue placement may promote chronic inflammatory condition because of the proinflammatory effect of the glue [6]. At our knowledge, there is no previous report on supravalvular aortic stenosis caused by double teflon felt at proximal anastomosis site for chronic aneurysm. It is possible that, especially in the female gender in whom the native aorta may be smaller, the use of double Teflon felt in
anastomosis techniques, with the healing process at the level of the inner felt, may result in some relative stenosis. Our patient was treated with the removal of the inside Teflon felt with the prompt disappearance of the peak gradient at the postoperative echocardiogram control. This case highlights this very rare late complication of aortic surgery and it might mean that when the native aorta is small (e.g. young patients, women) the reinforcement of the anastomosis by double teflon felt might produce aortic stenosis at the anastomotic site. In these cases only external Teflon felt may be sufficient to reinforce the anastomosis. Furthermore, careful and regular imaging of patients with prosthetic grafts of the aorta is mandatory to detect early and late complications.
References [1] Di Bartolomeo R, Di Marco L, Armaro A, et al. Treatment of complex disease of the thoracic aorta: the frozen elephant trunk technique with the E-vita open prosthesis. Eur J Cardiothoracic Surg 2009;35(4):671–5. [2] Turley AJ, Dark J, Adams PC. Acquired supravalvular aortic stenosis: a late complication of replacement of the ascending aorta. Surg 2008;34:690–2. [3] Cianciulli TF, Fairman EB, Saccheri MC, Llanos Dethinne SD, Prezioso HA. Acute supravalvular aortic stenosis following the replacement of the ascending aorta. Eur J Echocardiogr 2007;8:232–4. [4] Rose AG, Park SJ, Shumway SJ, Norton D, Miller LW. Acquired supravalvular aortic stenosis following heart transplantation: report of 2 cases. J Heart Lung Trasplant 2002;21:499–502. [5] Matsuura K, Ogino H, Minatoya K, Sasaki H. Aortic stenosis caused by the felt Teflon strip used in repair for acute aortic dissection. Interact Cardiovasc Thorac Surg 2004;3:41–3. [6] Kunihara T, Shiiya N, Matsuzaki K, Murashita T, Matsui Y. Recommendation for appropriate use of GRF glue in the operation for acute aortic dissection. Ann Thorac Cardiovasc Surg 2008;14:88–95.
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L. Di Marco et al. / International Journal of Cardiology 156 (2012) e62–e64
Fig. 2. A) Transthoracic echocardiography: continuous wave Doppler measurement in the ascending aorta at the proximal anastomosis site showed a broad systolic jet of high velocity (474 cm/s). Cardiac catheterization demonstrating a 90 mm Hg peak gradient through the proximal anastomosis between the sino-tubular junction and the Dacron prosthesis; B–C) Angio CT scan: severe size-reduction at the site of proximal anastomosis (minimum diameter: 11–12 mm). Distally to the E-vita prosthesis, increase of the aneurismal dilatation of the descending thoracic aorta.
Contents lists available at SciVerse ScienceDirect
International Journal of Cardiology journal homepage: www.elsevier.com/locate/ijcard
Letter to the Editor
Acquired aortic coarctation: A rare case of supraortic valve stenosis L. Di Marco a,⁎, D. Pacini a, R. Lorusso b, G.F. Pasini c, G. Zanini c, A. Pantaleo a, R. Di Bartolomeo a a b c
Cardiac Surgery Department, S. Orsola-Malpighi Hospital, University of Bologna, Bologna, Italy Cardiac Surgery Unit, Civic Hospital, Brescia, Italy Cardiology Unit, “La Memoria” Hospital, Gavardo, Italy
a r t i c l e
i n f o
Article history: Received 2 August 2011 Accepted 20 August 2011 Available online 13 September 2011 Keywords: Supravalvular stenosis Aortic valve Teflon felt Ascending aorta
Acquired supravalvular aortic stenosis (SVAS) is a very rare complication following cardiac surgery. Usually, SVAS is a congenital condition characterized by narrowing of the aorta close to its origin. It is either isolated or part of Williams syndrome. Moreover, isolated iatrogenic or acquired case of SVAS has been also reported. After an accurate review of the literature, this is one of the few cases of symptomatic supravalvular aortic stenosis after replacement of the ascending aorta. A 65-year-old Asian female was referred to our institution from other hospital with a diagnosis of supravalvular aortic stenosis. As cardiovascular risk factors she was hypertensive and diabetic. In 2010, we operated the patient for elective replacement of supracommissural ascending aorta, aortic arch and proximal portion of the descending thoracic aorta with frozen elephant trunk technique using a 28 mm E-vita Open Plus (Jotec GmbH, Germany) because of chronic aneurysm of the thoracic aorta. Our technique for complete replacement of the thoracic aorta with frozen elephant trunk has been already described [1].Once the graft was delivered in the proximal portion of the descending thoracic aorta and the aortic arch replaced, the Dacron graft was sutured to the previously prepared supracommissural ascending aorta with a double internal–external Teflon felt used to reinforce the suture. Postoperatively, the patient had an uneventful recovery, the angio-Computed Tomography (CT) scan showed a slight reduction in size of the ascending aorta lumen at the proximal site of the anastomosis and a complete exclusion of the aneurysmal dilatation distally to
⁎ Corresponding author at: Cardiac Surgery Department, University of Bologna, S. Orsola-Malpighi Hospital, Via Massarenti, 9, 40138 Bologna, Italy. Tel.: +39 0516363361; fax: +39 051345990. E-mail address: [email protected] (L. Di Marco). 0167-5273/$ – see front matter © 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijcard.2011.08.069
the E-vita prosthesis so she was discharged (Fig. 1). One year later, she was hospitalized for the onset of dyspnea on exertion and for occasional episodes of dizziness. Transthoracic echocardiography (TTE) showed left ventricular normal function (ejection fraction 56%), the aortic valve showed a mild aortic regurgitation and the tricuspid valve a moderate regurgitation with severe pulmonary hypertension (peak value 80 mm Hg).Doppler revealed a turbulent flow in the proximal portion of the ascending aorta with a pressure gradient between left ventricle and the aorta of 109 mm Hg. The patient underwent to transesophageal echocardiography (TEE) which revealed an ascending aorta acceleration flow at 3.7 cm from the aortic annulus with a persistence of transaortic supravalvular gradient of 93 mm Hg. The angio-CT scan control revealed a significative stenosis of the ascending aorta at the proximal suture with Dacron portion of the E-vita prosthesis at 3 cm from the aortic annulus with a severe reduction of the lumen at 1.1 cm. It also showed a type 1 endoleak distally to the E-vita graft with an aneurysm of the distal portion of the descending thoracic aorta of 5.9 cm. In the light of this instrumental finding, the patient was then referred at our Institution with a diagnosis of supravalvular aortic stenosis. On admission, the patient was asymptomatic. An electrocardiogram showed left ventricular hypertrophy.TTE confirmed severe iatrogenic supravalvular stenosis attributable to surgical material at 3 cm from the valvular plane with a peak gradient pressure of 90 mm Hg (Fig. 2A). The patient repeated the angio-CT scan which confirmed a severe caliber-shrinkage of the ascending aorta at proximal anastomosis site (minimum diameter: 11–12 mm) and an increase of the aneurismal dilatation of the descending thoracic aorta distally to the E-vita prosthesis (Fig. 2B–C). The patient underwent reoperation. The proximal anastomosis was opened, and we detected that the inner teflon felt created a supravalvular labrum causing the supraortic valve stenosis. It was then removed. The proximal supracoronary aorta was suitably shaped in oblique fashion in order to make wider anastomosis. The proximal anastomosis between the graft and the ascending aorta was then completed and reinforced with the only external Teflon felt. Following an uneventful recovery period, the patient underwent to TTE control which showed the absence of the gradient at the site of proximal anastomosis. The patient was then discharged. Postoperative bleeding represents one of the main problem of aortic surgery. To reduce its incidence, various techniques have been developed. The use of external or double Teflon felt, internal and external, at the site of anastomosis, represents one of these. A double Teflon felt strip may be used outside and inside the aorta, and fixed with a horizontal mattress suture. Care must be taken when the
L. Di Marco et al. / International Journal of Cardiology 156 (2012) e62–e64
e63
Fig. 1. Angio CT scan; A–B): The CT scan shows a slight size-reduction at the proximal site of the anastomosis (diameter: 23 mm) between native ascending aorta and Dacron prosthesis. Distally to the E-vita prosthesis, complete exclusion of the aneurismal dilatation of the descending thoracic aorta.
suture is tied in order to avoid the excessive narrowing of the aorta: in fact, if the mattress suture is too much pulled, the lumen of the aorta may be narrowed, especially if the native aorta is small. The present report shows a very unusual complication following ascending aorta replacement, presenting as supravalvular aortic stenosis and demonstrated by echocardiographic finding of a flow acceleration with a gradient pressure at the level of the proximal anastomosis between supracommissural portion of the ascending aorta and proximal Dacron portion of the E-vita prosthesis. Usually, SVAS is a congenital condition and it is strongly associated with Williams syndrome, a defect involving the elastin gene, although isolated iatrogenic or acquired case of SVAS have been previously reported [2,3]. SVAS may result from the kinking caused by redundant donor ascending aorta tissue following heart transplantation or from peri-aortic pseudoaneurysm compressing the aortic graft [3,4].The present report describes a case of iatrogenic stenosis of the supracoronary ascending aorta after replacement for chronic aneurysm, due to the healing process at the level of the proximal anastomosis which caused a stiffening of the inner felt with consequent severe reduction of the ascending aorta lumen at the sinotubular junction. Previous authors described supraaortic stenosis originating from double Teflon felt associated with biological glue used to reinforce the proximal anastomosis in a case of type A aortic dissection [5]. As they pointed out, the Teflon felt used to reinforce the dissection following glue placement may promote chronic inflammatory condition because of the proinflammatory effect of the glue [6]. At our knowledge, there is no previous report on supravalvular aortic stenosis caused by double teflon felt at proximal anastomosis site for chronic aneurysm. It is possible that, especially in the female gender in whom the native aorta may be smaller, the use of double Teflon felt in
anastomosis techniques, with the healing process at the level of the inner felt, may result in some relative stenosis. Our patient was treated with the removal of the inside Teflon felt with the prompt disappearance of the peak gradient at the postoperative echocardiogram control. This case highlights this very rare late complication of aortic surgery and it might mean that when the native aorta is small (e.g. young patients, women) the reinforcement of the anastomosis by double teflon felt might produce aortic stenosis at the anastomotic site. In these cases only external Teflon felt may be sufficient to reinforce the anastomosis. Furthermore, careful and regular imaging of patients with prosthetic grafts of the aorta is mandatory to detect early and late complications.
References [1] Di Bartolomeo R, Di Marco L, Armaro A, et al. Treatment of complex disease of the thoracic aorta: the frozen elephant trunk technique with the E-vita open prosthesis. Eur J Cardiothoracic Surg 2009;35(4):671–5. [2] Turley AJ, Dark J, Adams PC. Acquired supravalvular aortic stenosis: a late complication of replacement of the ascending aorta. Surg 2008;34:690–2. [3] Cianciulli TF, Fairman EB, Saccheri MC, Llanos Dethinne SD, Prezioso HA. Acute supravalvular aortic stenosis following the replacement of the ascending aorta. Eur J Echocardiogr 2007;8:232–4. [4] Rose AG, Park SJ, Shumway SJ, Norton D, Miller LW. Acquired supravalvular aortic stenosis following heart transplantation: report of 2 cases. J Heart Lung Trasplant 2002;21:499–502. [5] Matsuura K, Ogino H, Minatoya K, Sasaki H. Aortic stenosis caused by the felt Teflon strip used in repair for acute aortic dissection. Interact Cardiovasc Thorac Surg 2004;3:41–3. [6] Kunihara T, Shiiya N, Matsuzaki K, Murashita T, Matsui Y. Recommendation for appropriate use of GRF glue in the operation for acute aortic dissection. Ann Thorac Cardiovasc Surg 2008;14:88–95.
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L. Di Marco et al. / International Journal of Cardiology 156 (2012) e62–e64
Fig. 2. A) Transthoracic echocardiography: continuous wave Doppler measurement in the ascending aorta at the proximal anastomosis site showed a broad systolic jet of high velocity (474 cm/s). Cardiac catheterization demonstrating a 90 mm Hg peak gradient through the proximal anastomosis between the sino-tubular junction and the Dacron prosthesis; B–C) Angio CT scan: severe size-reduction at the site of proximal anastomosis (minimum diameter: 11–12 mm). Distally to the E-vita prosthesis, increase of the aneurismal dilatation of the descending thoracic aorta.