Analysis of risk factors for surgical treatment of congenital pulmonary venous stenosis combined with congenital heart disease_Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Authors：

LV Xiaodong ¹ , GAO Zhan ² , TENG Xiao ¹ , GAO Qiang ² , HUA Zhongdong ¹ , YAN Jun ¹ , ZHANG Zewei ² ,  LI Shoujun ¹

1. Pediatric Cardiac Surgery Center, Fuwai Hospital, Beijing Union Medical University, Chinese Academy of Medical Sciences, Beijing, 100037, P.R.China;
2. Department of Cardiovascular Surgery, Children's Hospital, School of Medicine, Zhejiang University, Hangzhou, 310000, P.R.China;

Corresponding author：

LI Shoujun, Email: drshoujunlifw@163.com

Keywords：

Pulmonary vein stenosis; congenital heart disease; pulmonary hypertension; risk factor; surgery

DOI：

10.7507/1007-4848.202008023

Video：

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Abstract Full text Figures/Tables Video References Cited by

Objective To explore risk factors associated with mortality and restenosis after the surgery for congenital pulmonary venous stenosis (CPVS) combined with congenital heart disease.Methods From May 2007 to August 2019, 58 patients received surgical relief of CPVS combined with congenital heart disease, including 24 males and 34 females, aged 17.2±26.3 months, weighing 8.8±8.2 kg. Endpoints were death and restenosis, and the risk factors were analyzed. A univariate and multivariate risk analyses were performed.Results Preoperative pulmonary venous stenosis severity score (PVSSS) was 4.5±2.7. Average pulmonary vein counts with CPVS was 1.9±1.0. There were 2 (3.4%) early deaths. The mean follow-up time was 2-145 (49.8±40.0) months. The 1-, 2-, 3- and 5-year overall survival rates were 86.7%, 81.3%, 78.5% and 73.6%, respectively, and the pulmonary venous restenosis-free rates were 79.6%, 68.5%, 68.5% and 68.5%, respectively. Preterm birth was an independent risk factor for mortality. The pulmonary venous peak flow rate ≥1.2 m/s at discharge was an independent risk factor for mortality and restenosis.Conclusion The prognosis of CPVS is still poor. Postoperative residual stenosis at discharge is an independent risk factor for death and restenosis.

Citation： LV Xiaodong, GAO Zhan, TENG Xiao, GAO Qiang, HUA Zhongdong, YAN Jun, ZHANG Zewei, LI Shoujun. Analysis of risk factors for surgical treatment of congenital pulmonary venous stenosis combined with congenital heart disease. Chinese Journal of Clinical Thoracic and Cardiovascular Surgery, 2021, 28(12): 1455-1460. doi: 10.7507/1007-4848.202008023 Copy

1.	Kalfa D, Belli E, Bacha E, et al. Primary pulmonary vein stenosis: Outcomes, risk factors, and severity score in a multicentric study. Ann Thorac Surg, 2017, 104(1): 182-189.
2.	Shi G, Zhu Z, Chen H, et al. Surgical repair for primary pulmonary vein stenosis: Single-institution, midterm follow-up. J Thorac Cardiovasc Surg, 2015, 150(1): 181-188.
3.	Rosenblum JM, Altin HF, Gillespie SE, et al. Management outcomes of primary pulmonary vein stenosis. J Thorac Cardiovasc Surg, 2020, 159(3): 1029-1036.
4.	Abe Y, Sumitomo N, Ayusawa M, et al. Congenital multiple pulmonary vein atresia and stenosis in an infant. Pediatr Int, 2018, 60(10): 976-978.
5.	Mahgoub L, Kaddoura T, Kameny AR, et al. Pulmonary vein stenosis of ex-premature infants with pulmonary hypertension and bronchopulmonary dysplasia, epidemiology, and survival from a multicenter cohort. Pediatr Pulmonol, 2017, 52(8): 1063-1070.
6.	DiLorenzo MP, Santo A, Rome JJ, et al. Pulmonary vein stenosis: Outcomes in children with congenital heart disease and prematurity. Semin Thorac Cardiovasc Surg, 2019, 31(2): 266-273.
7.	Charlagorla P, Becerra D, Patel PM, et al. Congenital pulmonary vein stenosis: Encouraging mid-term outcome. Pediatr Cardiol, 2016, 37(1): 125-130.
8.	王建德, 李建蓉. 肺静脉狭窄的诊治及进展. 中华医学超声杂志(电子版), 2010, 7(9): 1560-1563.
9.	Lo Rito M, Gazzaz T, Wilder TJ, et al. Pulmonary vein stenosis: Severity and location predict survival after surgical repair. J Thorac Cardiovasc Surg, 2016, 151(3): 657-666.
10.	Callahan R, Kieran MW, Baird CW, et al. Adjunct targeted biologic inhibition agents to treat aggressive multivessel intraluminal pediatric pulmonary vein stenosis. J Pediatr, 2018, 198: 29-35.
11.	Balasubramanian S, Rehman M, Gauvreau K, et al. Bilateral disease and early age at presentation are associated with shorter survival in patients with congenital heart disease and intraluminal pulmonary vein stenosis. Congenit Heart Dis, 2012, 7(4): 378-386.
12.	Altit G, Bhombal S, Hopper RK, et al. Death or resolution: The "natural history" of pulmonary hypertension in bronchopulmonary dysplasia. J Perinatol, 2019, 39(3): 415-425.
13.	Kalfa D, Belli E, Bacha E, et al. Outcomes and prognostic factors for postsurgical pulmonary vein stenosis in the current era. J Thorac Cardiovasc Surg, 2018, 156(1): 278-286.
14.	Nasr VG, Callahan R, Wichner Z, et al. Intraluminal pulmonary vein stenosis in children: A "new" lesion. Anesth Analg, 2019, 129(1): 27-40.
15.	Vanderlaan RD, Caldarone CA. Sutureless repair and postoperative residual stenosis: "Never leave with a lesion". J Thorac Cardiovasc Surg, 2018, 156(1): 287-288.
16.	Zhu J, Ide H, Fu YY, et al. Losartan ameliorates "upstream" pulmonary vein vasculopathy in a piglet model of pulmonary vein stenosis. J Thorac Cardiovasc Surg, 2014, 148(6): 2550-2557.

1. Kalfa D, Belli E, Bacha E, et al. Primary pulmonary vein stenosis: Outcomes, risk factors, and severity score in a multicentric study. Ann Thorac Surg, 2017, 104(1): 182-189.
2. Shi G, Zhu Z, Chen H, et al. Surgical repair for primary pulmonary vein stenosis: Single-institution, midterm follow-up. J Thorac Cardiovasc Surg, 2015, 150(1): 181-188.
3. Rosenblum JM, Altin HF, Gillespie SE, et al. Management outcomes of primary pulmonary vein stenosis. J Thorac Cardiovasc Surg, 2020, 159(3): 1029-1036.
4. Abe Y, Sumitomo N, Ayusawa M, et al. Congenital multiple pulmonary vein atresia and stenosis in an infant. Pediatr Int, 2018, 60(10): 976-978.
5. Mahgoub L, Kaddoura T, Kameny AR, et al. Pulmonary vein stenosis of ex-premature infants with pulmonary hypertension and bronchopulmonary dysplasia, epidemiology, and survival from a multicenter cohort. Pediatr Pulmonol, 2017, 52(8): 1063-1070.
6. DiLorenzo MP, Santo A, Rome JJ, et al. Pulmonary vein stenosis: Outcomes in children with congenital heart disease and prematurity. Semin Thorac Cardiovasc Surg, 2019, 31(2): 266-273.
7. Charlagorla P, Becerra D, Patel PM, et al. Congenital pulmonary vein stenosis: Encouraging mid-term outcome. Pediatr Cardiol, 2016, 37(1): 125-130.
8. 王建德, 李建蓉. 肺静脉狭窄的诊治及进展. 中华医学超声杂志(电子版), 2010, 7(9): 1560-1563.
9. Lo Rito M, Gazzaz T, Wilder TJ, et al. Pulmonary vein stenosis: Severity and location predict survival after surgical repair. J Thorac Cardiovasc Surg, 2016, 151(3): 657-666.
10. Callahan R, Kieran MW, Baird CW, et al. Adjunct targeted biologic inhibition agents to treat aggressive multivessel intraluminal pediatric pulmonary vein stenosis. J Pediatr, 2018, 198: 29-35.
11. Balasubramanian S, Rehman M, Gauvreau K, et al. Bilateral disease and early age at presentation are associated with shorter survival in patients with congenital heart disease and intraluminal pulmonary vein stenosis. Congenit Heart Dis, 2012, 7(4): 378-386.
12. Altit G, Bhombal S, Hopper RK, et al. Death or resolution: The "natural history" of pulmonary hypertension in bronchopulmonary dysplasia. J Perinatol, 2019, 39(3): 415-425.
13. Kalfa D, Belli E, Bacha E, et al. Outcomes and prognostic factors for postsurgical pulmonary vein stenosis in the current era. J Thorac Cardiovasc Surg, 2018, 156(1): 278-286.
14. Nasr VG, Callahan R, Wichner Z, et al. Intraluminal pulmonary vein stenosis in children: A "new" lesion. Anesth Analg, 2019, 129(1): 27-40.
15. Vanderlaan RD, Caldarone CA. Sutureless repair and postoperative residual stenosis: "Never leave with a lesion". J Thorac Cardiovasc Surg, 2018, 156(1): 287-288.
16. Zhu J, Ide H, Fu YY, et al. Losartan ameliorates "upstream" pulmonary vein vasculopathy in a piglet model of pulmonary vein stenosis. J Thorac Cardiovasc Surg, 2014, 148(6): 2550-2557.

Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Analysis of risk factors for surgical treatment of congenital pulmonary venous stenosis combined with congenital heart disease

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