C. Pizarro et al. / European Journal of Cardio-Thoracic Surgery In terms of the evolution of the strategy, I think that as we move along, we have learned that different people have different expectations and obviously this is not a curative procedure. The disappointing fact was that we were using this procedure for certain conditions which will not resolve and therefore the risk will remain. I think that there are many other centres which have published data regarding their patient selection as to which patients they choose to employ this strategy for management early on, with the idea that the patient will improve and could have a better outcome. But the reality is that it seems like you shift rather than eliminate the risks along the staging process. And I think it has become clear that if you have a reversible condition that may improve, that’s a perfect scenario for this. For example, patients who had interrupted aortic arch with VSD, posterior malalignment type with critical sub AS, and AS with a trachea-oesophageal fistula, or duodenal atresia that needed an operation. A quick hybrid was done in those patients, then they went for those interventions, they recovered, and then we did a complete biventricular repair and it worked out great.
1001
Some patients with very poor prognosis have this as a palliative intervention because the parents want to take the child home knowing that the child is going to die, rather than staying in the hospital. And we have done a couple for those indications. So I think the indication has evolved greatly over a span of about ten years. Dr Anderson: And so with respect to aortic atresia, I mean the obvious problem is occlusion of the isthmus. Have you any experience of placing a stent in that either at the time or at a later date? Dr Pizarro: No. Initially we tried to balloon a couple of patients without a great deal of success, and now we follow some of the recommendations from the group in Columbus to move early on to a Stage I Norwood in those cases when there is either evidence of ventricular dysfunction or critical narrowing that results in progressive increase in velocity in the retrograde flow. And if you looked at the results in these patients, these three patients post Norwood, they were all patients who had an indication to do something sooner rather than later because they were in trouble, and obviously they didn’t do well.
EDITORIAL COMMENT
European Journal of Cardio-Thoracic Surgery 47 (2015) 1001–1002 doi:10.1093/ejcts/ezu345 Advance Access publication 24 November 2014
The questionable role of the hybrid procedure Amir-Reza Hosseinpour* The Heart Unit, University Hospitals Virgen del Rocio, Seville, Spain * Corresponding author. Department of Cardiac Surgery, University Hospitals Virgen del Rocio, Avenida Manuel Siurot s/n, 41013 Seville, Spain. fax: +34-955-012359; e-mail: [email protected] (A-R. Hosseinpour).
Keywords: Hybrid procedure • Norwood procedure • Hypoplastic left heart syndrome
The hybrid procedure was first proposed in 1993, and again in 2002, as an alternative to the Norwood procedure for hypoplastic left heart syndrome, in order to avoid complex cardiac surgery in the neonatal period with all its possible neuro-developmental sequelae [1, 2]. It became popular, however, with the idea that it may prove useful for a subgroup of patients at very high risk of mortality after the Norwood procedure, despite the remarkable reduction in the overall mortality of this operation [3, 4]. These are babies with low weight and/or aortic atresia. The hybrid procedure has been used increasingly for over a decade, without being confined to high-risk cases. Nevertheless, it has failed to show its superiority to the Norwood procedure [5]. This prompted Pizarro et al. to dissect out and define the major risk factors for this procedure, hoping to improve its results by case selection [6]. They show that the two major risk factors for mortality in the Norwood procedure (low weight and aortic atresia) also apply to the hybrid approach, with no survivors when both risk factors coexist. This finding is very important; it makes case selection problematic and paradoxical, since excluding these babies defeats a major purpose of the hybrid procedure. Indeed, case selection here discriminates against the very patients this procedure was hoped to serve. This questions the utility of this approach altogether. For the hybrid procedure to be useful when compared with the Norwood procedure, it must either achieve better neuro-
developmental outcome, or lower overall mortality, or better outcome in high-risk cases. Its neuro-developmental outcome has already disappointed in one study at 1-year follow-up, although more work is needed to clarify this [7]. Lower overall mortality is not achieved [5]. Now, it turns out that it cannot help the high-risk babies either [6]. Furthermore, the hybrid procedure is actually worse than the Norwood procedure in at least one important aspect—distortion of the branch pulmonary arteries by banding, as evidenced by the high rate of reinterventions on these vessels [8–10]. This is a very significant, yet under-emphasized, drawback of the hybrid procedure, since these patients are being prepared for an eventual Fontan which may be jeopardized by pulmonary arterial distortion. So, what does the hybrid procedure offer? It has certainly failed to live up to the original expectations. In addition, its important disadvantage with respect to the risk of pulmonary arterial distortion has been brought to light. However, as Pizarro et al. highlight, it may have found another role as a temporizing measure for cases where an eventual biventricular repair is planned [6]. Although such a temporizing measure may also be achieved with the Norwood procedure, subsequent biventricular repair is shorter and easier (and presumably safer) if the first procedure was the hybrid approach. Also, the concern regarding pulmonary arterial distortion is less significant since a biventricular repair is
CONGENITAL
Cite this article as: Hosseinpour A-R. The questionable role of the hybrid procedure. Eur J Cardiothorac Surg 2015;47:1001–2.
1002
C. Pizarro et al. / European Journal of Cardio-Thoracic Surgery
more forgiving in this respect. This role may turn out to be the main application of this procedure. Conflict of interest: none declared.
REFERENCES [1] Gibbs JL, Wren C, Watterson KG, Hunter S, Hamilton JR. Stenting of the arterial duct combined with banding of the pulmonary arteries and atrial septectomy or septostomy: a new approach to palliation for the hypoplastic left heart syndrome. Br Heart J 1993;69:551–5. [2] Akintuerk H, Michel-Behnke I, Valeske K, Mueller M, Thul J, Bauer J et al. Stenting of the arterial duct and banding of the pulmonary arteries: basis for combined Norwood stage I and II repair in hypoplastic left heart. Circulation 2002;105:1099–103. [3] Bacha EA, Daves S, Hardin J, Abdulla RI, Anderson J, Kahana M et al. Single-ventricle palliation for high-risk neonates: the emergence of an alternative hybrid stage I strategy. J Thorac Cardiovasc Surg 2006;131:163–71. [4] Pizarro C, Derby CD, Baffa JM, Murdison KA, Radtke WA. Improving the outcome of high-risk neonates with hypoplastic left heart syndrome:
hybrid procedure or conventional surgical palliation? Eur J Cardiothorac Surg 2008;33:613–8. [5] Photiadis J, Sinzobahamvya N, Hraska V, Asfour B. Does bilateral pulmonary banding in comparison to Norwood procedure improve outcome in neonates with hypoplastic left heart syndrome beyond second-stage palliation? A review of the current literature. Thorac Cardiovasc Surg 2012;60:181–8. [6] Pizarro C, Davies RR, Woodford E, Radtke WA. Improving early outcomes following hybrid procedure for patients with single ventricle and systemic outflow obstruction: defining risk factors. Eur J Cardiothorac Surg 2015;47: 995–1001. [7] Knirsch W, Liamlahi R, Hug MI, Hoop R, von Rhein M, Prêtre R et al. Mortality and neurodevelopmental outcome at 1 year of age comparing hybrid and Norwood procedures. Eur J Cardiothorac Surg 2012;42:33–9. [8] Baba K, Kotani Y, Chetan D, Chaturvedi RR, Lee KJ, Benson LN et al. Hybrid versus Norwood strategies for single ventricle palliation. Circulation 2012; 126:S123–131. [9] Davies RR, Radtke WA, Klenk D, Pizarro C. Bilateral pulmonary arterial banding results in an increased need for subsequent pulmonary artery interventions. J Thorac Cardiovasc Surg 2014;147:706–12. [10] Dave H, Rosser B, Knirsch W, Hübler M, Prêtre R, Kretschmar O. Hybrid approach for hypoplastic left heart syndrome and its variants: the fate of the pulmonary arteries. Eur J Cardiothorac Surg 2014;46:14–9.
1001
Some patients with very poor prognosis have this as a palliative intervention because the parents want to take the child home knowing that the child is going to die, rather than staying in the hospital. And we have done a couple for those indications. So I think the indication has evolved greatly over a span of about ten years. Dr Anderson: And so with respect to aortic atresia, I mean the obvious problem is occlusion of the isthmus. Have you any experience of placing a stent in that either at the time or at a later date? Dr Pizarro: No. Initially we tried to balloon a couple of patients without a great deal of success, and now we follow some of the recommendations from the group in Columbus to move early on to a Stage I Norwood in those cases when there is either evidence of ventricular dysfunction or critical narrowing that results in progressive increase in velocity in the retrograde flow. And if you looked at the results in these patients, these three patients post Norwood, they were all patients who had an indication to do something sooner rather than later because they were in trouble, and obviously they didn’t do well.
EDITORIAL COMMENT
European Journal of Cardio-Thoracic Surgery 47 (2015) 1001–1002 doi:10.1093/ejcts/ezu345 Advance Access publication 24 November 2014
The questionable role of the hybrid procedure Amir-Reza Hosseinpour* The Heart Unit, University Hospitals Virgen del Rocio, Seville, Spain * Corresponding author. Department of Cardiac Surgery, University Hospitals Virgen del Rocio, Avenida Manuel Siurot s/n, 41013 Seville, Spain. fax: +34-955-012359; e-mail: [email protected] (A-R. Hosseinpour).
Keywords: Hybrid procedure • Norwood procedure • Hypoplastic left heart syndrome
The hybrid procedure was first proposed in 1993, and again in 2002, as an alternative to the Norwood procedure for hypoplastic left heart syndrome, in order to avoid complex cardiac surgery in the neonatal period with all its possible neuro-developmental sequelae [1, 2]. It became popular, however, with the idea that it may prove useful for a subgroup of patients at very high risk of mortality after the Norwood procedure, despite the remarkable reduction in the overall mortality of this operation [3, 4]. These are babies with low weight and/or aortic atresia. The hybrid procedure has been used increasingly for over a decade, without being confined to high-risk cases. Nevertheless, it has failed to show its superiority to the Norwood procedure [5]. This prompted Pizarro et al. to dissect out and define the major risk factors for this procedure, hoping to improve its results by case selection [6]. They show that the two major risk factors for mortality in the Norwood procedure (low weight and aortic atresia) also apply to the hybrid approach, with no survivors when both risk factors coexist. This finding is very important; it makes case selection problematic and paradoxical, since excluding these babies defeats a major purpose of the hybrid procedure. Indeed, case selection here discriminates against the very patients this procedure was hoped to serve. This questions the utility of this approach altogether. For the hybrid procedure to be useful when compared with the Norwood procedure, it must either achieve better neuro-
developmental outcome, or lower overall mortality, or better outcome in high-risk cases. Its neuro-developmental outcome has already disappointed in one study at 1-year follow-up, although more work is needed to clarify this [7]. Lower overall mortality is not achieved [5]. Now, it turns out that it cannot help the high-risk babies either [6]. Furthermore, the hybrid procedure is actually worse than the Norwood procedure in at least one important aspect—distortion of the branch pulmonary arteries by banding, as evidenced by the high rate of reinterventions on these vessels [8–10]. This is a very significant, yet under-emphasized, drawback of the hybrid procedure, since these patients are being prepared for an eventual Fontan which may be jeopardized by pulmonary arterial distortion. So, what does the hybrid procedure offer? It has certainly failed to live up to the original expectations. In addition, its important disadvantage with respect to the risk of pulmonary arterial distortion has been brought to light. However, as Pizarro et al. highlight, it may have found another role as a temporizing measure for cases where an eventual biventricular repair is planned [6]. Although such a temporizing measure may also be achieved with the Norwood procedure, subsequent biventricular repair is shorter and easier (and presumably safer) if the first procedure was the hybrid approach. Also, the concern regarding pulmonary arterial distortion is less significant since a biventricular repair is
CONGENITAL
Cite this article as: Hosseinpour A-R. The questionable role of the hybrid procedure. Eur J Cardiothorac Surg 2015;47:1001–2.
1002
C. Pizarro et al. / European Journal of Cardio-Thoracic Surgery
more forgiving in this respect. This role may turn out to be the main application of this procedure. Conflict of interest: none declared.
REFERENCES [1] Gibbs JL, Wren C, Watterson KG, Hunter S, Hamilton JR. Stenting of the arterial duct combined with banding of the pulmonary arteries and atrial septectomy or septostomy: a new approach to palliation for the hypoplastic left heart syndrome. Br Heart J 1993;69:551–5. [2] Akintuerk H, Michel-Behnke I, Valeske K, Mueller M, Thul J, Bauer J et al. Stenting of the arterial duct and banding of the pulmonary arteries: basis for combined Norwood stage I and II repair in hypoplastic left heart. Circulation 2002;105:1099–103. [3] Bacha EA, Daves S, Hardin J, Abdulla RI, Anderson J, Kahana M et al. Single-ventricle palliation for high-risk neonates: the emergence of an alternative hybrid stage I strategy. J Thorac Cardiovasc Surg 2006;131:163–71. [4] Pizarro C, Derby CD, Baffa JM, Murdison KA, Radtke WA. Improving the outcome of high-risk neonates with hypoplastic left heart syndrome:
hybrid procedure or conventional surgical palliation? Eur J Cardiothorac Surg 2008;33:613–8. [5] Photiadis J, Sinzobahamvya N, Hraska V, Asfour B. Does bilateral pulmonary banding in comparison to Norwood procedure improve outcome in neonates with hypoplastic left heart syndrome beyond second-stage palliation? A review of the current literature. Thorac Cardiovasc Surg 2012;60:181–8. [6] Pizarro C, Davies RR, Woodford E, Radtke WA. Improving early outcomes following hybrid procedure for patients with single ventricle and systemic outflow obstruction: defining risk factors. Eur J Cardiothorac Surg 2015;47: 995–1001. [7] Knirsch W, Liamlahi R, Hug MI, Hoop R, von Rhein M, Prêtre R et al. Mortality and neurodevelopmental outcome at 1 year of age comparing hybrid and Norwood procedures. Eur J Cardiothorac Surg 2012;42:33–9. [8] Baba K, Kotani Y, Chetan D, Chaturvedi RR, Lee KJ, Benson LN et al. Hybrid versus Norwood strategies for single ventricle palliation. Circulation 2012; 126:S123–131. [9] Davies RR, Radtke WA, Klenk D, Pizarro C. Bilateral pulmonary arterial banding results in an increased need for subsequent pulmonary artery interventions. J Thorac Cardiovasc Surg 2014;147:706–12. [10] Dave H, Rosser B, Knirsch W, Hübler M, Prêtre R, Kretschmar O. Hybrid approach for hypoplastic left heart syndrome and its variants: the fate of the pulmonary arteries. Eur J Cardiothorac Surg 2014;46:14–9.
