Pulmonary artery reconstruction to repair infant isolated unilateral absence of pulmonary artery: A retrospective cohort study in a single center_Chinese Journal of Clinical Thoracic and Cardiovascular Surgery

Authors：

YAN Xinjian , CHEN Jimei , CEN Jianzheng , WEN Shusheng , XU Gang , CUI Hujun , LIU Xiaobing ,  ZHUANG Jian

1. Department of Pediatric Cardiac Surgery, Guangdong Provincial People's Hospital, Guangdong Provincial Academy of Medical Sciences, Guangdong Institute of Cardiovascular Disease, Guangdong South China Key Laboratory of Structural Cardiology, Guangzhou, 510080,P. R. China;

Corresponding author：

ZHUANG Jian, Email: zhuangjian5413@tom.com

Keywords：

Pulmonary artery reconstruction; unilateral absence of pulmonary artery; congenital heart diseases

DOI：

10.7507/1007-4848.202209001

Video：

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Objective To confirm the changes of pulmonary artery pressure, neo pulmonary artery stenosis and reoperation in children with unilateral absence of pulmonary artery (UAPA) undergoing pulmonary artery reconstruction. Methods The clinical data of the infants with UAPA undergoing pulmonary artery reconstruction in our hospital from February 19, 2019 to April 15, 2021 were analyzed. Changes in pulmonary artery pressure, neo pulmonary artery stenosis and reoperation were followed up. Results Finally 5 patients were collected, including 4 males and 1 female. The operation age ranged from 13 days to 2.7 years. Cardiac contrast-enhanced CT scans were performed in all children, and 2 patients underwent pulmonary vein wedge angiography to confirm the diagnosis and preoperative evaluation. Preoperative transthoracic echocardiography and intraoperative direct pulmonary arterial pressure measurement indicated that all 5 children had pulmonary hypertension, with a mean pulmonary arterial pressure of 31.3±16.0 mm Hg. Pulmonary arterial pressure decreased immediately after pulmonary artery reconstruction to 16.8±4.2 mm Hg. The mean follow-up time was 18.9±4.7 months. All 5 patients survived during the follow-up period, and 1 patient had neo pulmonary artery stenosis or even occlusion and was re-operated. Conclusion Pulmonary artery reconstruction can effectively alleviate the pulmonary hypertension in children with UAPA. The patency of the neo pulmonary artery should be closely followed up after surgery, and re-pulmonary angioplasty should be performed if necessary.

Citation： YAN Xinjian, CHEN Jimei, CEN Jianzheng, WEN Shusheng, XU Gang, CUI Hujun, LIU Xiaobing, ZHUANG Jian. Pulmonary artery reconstruction to repair infant isolated unilateral absence of pulmonary artery: A retrospective cohort study in a single center. Chinese Journal of Clinical Thoracic and Cardiovascular Surgery, 2024, 31(4): 600-605. doi: 10.7507/1007-4848.202209001 Copy

1.	Kim GB, Ban JE, Bae EJ, et al. Rehabilitation of pulmonary artery in congenital unilateral absence of intrapericardial pulmonary artery. J Thorac Cardiovasc Surg, 2011, 141(1): 171-178.
2.	Bockeria LA, Makhachev OA, Khiriev TKh, et al. Congenital isolated unilateral absence of pulmonary artery and variants of collateral blood supply of the ipsilateral lung. Interact Cardiovasc Thorac Surg, 2011, 12(3): 509-510.
3.	Wang P, Yuan L, Shi J, et al. Isolated unilateral absence of pulmonary artery in adulthood: A clinical analysis of 65 cases from a case series and systematic review. J Thorac Dis, 2017, 9(12): 4988-4996.
4.	Ten Harkel AD, Blom NA, Ottenkamp J. Isolated unilateral absence of a pulmonary artery: A case report and review of the literature. Chest, 2002, 122(4): 1471-1477.
5.	Li W, Ma L, Xia S, et al. Early single-stage surgical revascularization of pulmonary artery in unilateral absence of a pulmonary artery. J Cardiothorac Surg, 2021, 16(1): 80.
6.	Apostolopoulou SC, Kelekis NL, Brountzos EN, et al. "Absent" pulmonary artery in one adult and five pediatric patients: Imaging, embryology, and therapeutic implications. AJR Am J Roentgenol, 2002, 179(5): 1253-1260.
7.	Kruzliak P, Syamasundar RP, Novak M, et al. Unilateral absence of pulmonary artery: Pathophysiology, symptoms, diagnosis and current treatment. Arch Cardiovasc Dis, 2013, 106(8-9): 448-454.
8.	Welch K, Hanley F, Johnston T, et al. Isolated unilateral absence of right proximal pulmonary artery: Surgical repair and follow-up. Ann Thorac Surg, 2005, 79(4): 1399-1402.
9.	Raymond A, Pedretti E, Privitera G, et al. Neonatal diagnosis of isolated absence of the right pulmonary artery: A case report and review of the literature. Ital J Pediatr, 2018, 44(1): 27.
10.	Bouros D, Pare P, Panagou P, et al. The varied manifestation of pulmonary artery agenesis in adulthood. Chest, 1995, 108(3): 670-676.
11.	Koga H, Hidaka T, Miyako K, et al. Age-related clinical characteristics of isolated congenital unilateral absence of a pulmonary artery. Pediatr Cardiol, 2010, 31(8): 1186-1190.
12.	Steiropoulos P, Archontogeorgis K, Tzouvelekis A, et al. Unilateral pulmonary artery agenesis: A case series. Hippokratia, 2013, 17(1): 73-76.
13.	Pfefferkorn JR, Löser H, Pech G, et al. Absent pulmonary artery. A hint to its embryogenesis. Pediatr Cardiol, 1982, 3(4): 283-286.
14.	Murphy DN, Winlaw DS, Cooper SG, et al. Successful early surgical recruitment of the congenitally disconnected pulmonary artery. Ann Thorac Surg, 2004, 77(1): 29-35.
15.	Moreno-Cabral RJ, McNamara JJ, Reddy VJ, et al. Unilateral absent pulmonary artery: Surgical repair with a new technique. J Thorac Cardiovasc Surg, 1991, 102(3): 463-465.
16.	Hiramatsu T, Komori S, Okamura Y, et al. Surgical correction of isolated unilateral absence of right pulmonary artery. Heart Vessels, 2010, 25(4): 353-355.
17.	Kosaka Y, Kurosawa H, Hoshino S, et al. Surgery for unilateral absence of pulmonary artery using autologous tissue. Ann Thorac Surg, 2003, 76(4): 1281-1283.
18.	Krammoh EK, Bigras JL, Prsa M, et al. Therapeutic strategies in children with an isolated unilaterally absent proximal pulmonary artery. Pediatr Cardiol, 2010, 31(5): 607-610.
19.	Pourmoghadam KK, Schwartz MC, DeCampli WM, et al. Novel approach to construct absent intrapericardial branch pulmonary arteries. Ann Thorac Surg, 2015, 100(2): 738-740.
20.	von Stumm M, Biermann D, Reichenspurner H, et al. Autologous tissue technique to repair unilateral isolated absence of a pulmonary artery. World J Pediatr Congenit Heart Surg, 2021, 12(4): 547-559.
21.	Mery CM, Molina KM, Krishnamurthy R, et al. Pulmonary artery resuscitation for isolated ductal origin of a pulmonary artery. J Thorac Cardiovasc Surg, 2014, 148(5): 2235-2244.

1. Kim GB, Ban JE, Bae EJ, et al. Rehabilitation of pulmonary artery in congenital unilateral absence of intrapericardial pulmonary artery. J Thorac Cardiovasc Surg, 2011, 141(1): 171-178.
2. Bockeria LA, Makhachev OA, Khiriev TKh, et al. Congenital isolated unilateral absence of pulmonary artery and variants of collateral blood supply of the ipsilateral lung. Interact Cardiovasc Thorac Surg, 2011, 12(3): 509-510.
3. Wang P, Yuan L, Shi J, et al. Isolated unilateral absence of pulmonary artery in adulthood: A clinical analysis of 65 cases from a case series and systematic review. J Thorac Dis, 2017, 9(12): 4988-4996.
4. Ten Harkel AD, Blom NA, Ottenkamp J. Isolated unilateral absence of a pulmonary artery: A case report and review of the literature. Chest, 2002, 122(4): 1471-1477.
5. Li W, Ma L, Xia S, et al. Early single-stage surgical revascularization of pulmonary artery in unilateral absence of a pulmonary artery. J Cardiothorac Surg, 2021, 16(1): 80.
6. Apostolopoulou SC, Kelekis NL, Brountzos EN, et al. "Absent" pulmonary artery in one adult and five pediatric patients: Imaging, embryology, and therapeutic implications. AJR Am J Roentgenol, 2002, 179(5): 1253-1260.
7. Kruzliak P, Syamasundar RP, Novak M, et al. Unilateral absence of pulmonary artery: Pathophysiology, symptoms, diagnosis and current treatment. Arch Cardiovasc Dis, 2013, 106(8-9): 448-454.
8. Welch K, Hanley F, Johnston T, et al. Isolated unilateral absence of right proximal pulmonary artery: Surgical repair and follow-up. Ann Thorac Surg, 2005, 79(4): 1399-1402.
9. Raymond A, Pedretti E, Privitera G, et al. Neonatal diagnosis of isolated absence of the right pulmonary artery: A case report and review of the literature. Ital J Pediatr, 2018, 44(1): 27.
10. Bouros D, Pare P, Panagou P, et al. The varied manifestation of pulmonary artery agenesis in adulthood. Chest, 1995, 108(3): 670-676.
11. Koga H, Hidaka T, Miyako K, et al. Age-related clinical characteristics of isolated congenital unilateral absence of a pulmonary artery. Pediatr Cardiol, 2010, 31(8): 1186-1190.
12. Steiropoulos P, Archontogeorgis K, Tzouvelekis A, et al. Unilateral pulmonary artery agenesis: A case series. Hippokratia, 2013, 17(1): 73-76.
13. Pfefferkorn JR, Löser H, Pech G, et al. Absent pulmonary artery. A hint to its embryogenesis. Pediatr Cardiol, 1982, 3(4): 283-286.
14. Murphy DN, Winlaw DS, Cooper SG, et al. Successful early surgical recruitment of the congenitally disconnected pulmonary artery. Ann Thorac Surg, 2004, 77(1): 29-35.
15. Moreno-Cabral RJ, McNamara JJ, Reddy VJ, et al. Unilateral absent pulmonary artery: Surgical repair with a new technique. J Thorac Cardiovasc Surg, 1991, 102(3): 463-465.
16. Hiramatsu T, Komori S, Okamura Y, et al. Surgical correction of isolated unilateral absence of right pulmonary artery. Heart Vessels, 2010, 25(4): 353-355.
17. Kosaka Y, Kurosawa H, Hoshino S, et al. Surgery for unilateral absence of pulmonary artery using autologous tissue. Ann Thorac Surg, 2003, 76(4): 1281-1283.
18. Krammoh EK, Bigras JL, Prsa M, et al. Therapeutic strategies in children with an isolated unilaterally absent proximal pulmonary artery. Pediatr Cardiol, 2010, 31(5): 607-610.
19. Pourmoghadam KK, Schwartz MC, DeCampli WM, et al. Novel approach to construct absent intrapericardial branch pulmonary arteries. Ann Thorac Surg, 2015, 100(2): 738-740.
20. von Stumm M, Biermann D, Reichenspurner H, et al. Autologous tissue technique to repair unilateral isolated absence of a pulmonary artery. World J Pediatr Congenit Heart Surg, 2021, 12(4): 547-559.
21. Mery CM, Molina KM, Krishnamurthy R, et al. Pulmonary artery resuscitation for isolated ductal origin of a pulmonary artery. J Thorac Cardiovasc Surg, 2014, 148(5): 2235-2244.

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Pulmonary artery reconstruction to repair infant isolated unilateral absence of pulmonary artery: A retrospective cohort study in a single center

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