支气管肺发育不良极早产儿合并肺静脉狭窄2例报告
Pulmonary vein stenosis in extremely premature infants with bronchopulmonary dysplasia: a report of two cases
Received date: 2022-05-11
Online published: 2023-04-07
目的 探讨支气管肺发育不良极早产儿合并肺静脉狭窄（PVS）的临床特征和治疗结局。方法 回顾性分析两例支气管肺发育不良合并PVS极早产儿的临床资料，并复习相关文献。结果 两例极早产儿，胎龄分别为30+1周和28+2周，均为小于胎龄儿，诊断为支气管肺发育不良伴肺动脉高压。病例1生后4月20天发现左侧PVS，经药物治疗无效后死亡。病例2生后3月发现左侧PVS，生后5月15天右上肺静脉也狭窄，行肺静脉球囊扩张介入术后肺动脉压下降出院；1月余后，患儿肺高压反复，心胸外科行肺静脉狭窄纠治术，术后肺静脉再次狭窄，家属放弃治疗后死亡。结论 合并支气管肺发育不良伴肺动脉高压的极早产儿，动态复查心彩超时应特别注意评估肺静脉情况。PVS患儿扩肺血管药物治疗无效时，可尝试介入或外科手术干预，但总体死亡率较高。
王丽平 , 尤优 , 殷张华 , 王依闻 , 陈笋 , 夏红萍 . 支气管肺发育不良极早产儿合并肺静脉狭窄2例报告[J]. 临床儿科杂志, 2023 , 41(4) : 289 -293 . DOI: 10.12372/jcp.2023.22e0665
Objective To investigate the clinical characteristics and treatment of pulmonary vein stenosis (PVS) in extremely premature infants with bronchopulmonary dysplasia. Methods The clinical characteristics of PVS in two extremely premature infants with bronchopulmonary dysplasia were retrospectively analyzed and the related literatures were reviewed. Results The two infants were both small for gestational age and the gestational age was 30+1 weeks and 28+2 weeks, respectively. They were diagnosed with bronchopulmonary dysplasia with pulmonary hypertension. Case 1 had left PVS at 4 months and 20 days after birth, and he died after ineffective drug treatment. Case 2 had left PVS at 3 months after birth, and right upper PVS was found at 5 months and 15 days after birth. She received pulmonary vein balloon dilation therapy and pulmonary hypertension improved. More than a month after discharge, PVS relapsed and she received PVS correction operation. However, pulmonary vein turned stenosis again, the parents gave up further treatment and she died later. Conclusions Echocardiography should be performed in extremely premature infants with bronchopulmonary dysplasia and pulmonary hypertension, and pulmonary vein need to be evaluated carefully. When vasodilator drug treatment is ineffective, intervention or surgical operation could be considered. However, the overall mortality rate is high.
| [1] | Vanderlaan RD, Rome J, Hirsch R, et al. Pulmonary vein stenosis: Treatment and challenges[J]. J Thorac Cardiovasc Surg, 2021, 161(6): 2169-2176. |
| [2] | Laux D, Rocchisani MA, Boudjemline Y, et al. Pulmonary hypertension in the preterm infant with chronic lung disease can be caused by pulmonary vein stenosis: a must-know entity[J]. Pediatr Cardiol, 2016, 37(2): 313-321. |
| [3] | Backes CH, Nealon E, Armstrong AK, et al. Pulmonary vein stenosis in infants: a systematic review, meta-analysis, and meta-regression[J]. J Pediatr, 2018, 198: 36-45. |
| [4] | Frank DB, Levy PT, Stiver CA, et al. Primary pulmonary vein stenosis during infancy: state of the art review[J]. J Perinatol, 2021, 41(7): 1528-1539. |
| [5] | Higgins RD, Jobe AH, Koso-Thomas M, et al. Bronchopulmonary dysplasia: executive summary of a workshop[J]. J Pediatr, 2018, 197: 300-308. |
| [6] | 丁瑛雪. 早产儿支气管肺发育不良的表型演变[J]. 临床儿科杂志, 2022, 40(6): 407-412. |
| [7] | Latson LA, Prieto LR. Congenital and acquired pulmonary vein stenosis[J]. Circulation, 2007, 115(1): 103-108. |
| [8] | Jadcherla AV, Backes CH, Cua CL, et al. Primary pulmonary vein stenosis: a new look at a rare but challenging disease[J]. Neoreviews, 2021, 22(5): e296-e308. |
| [9] | 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[J]. Pediatr Pulmonol, 2017, 52(8): 1063-1670. |
| [10] | Chakrabarti S, Mittal R, Gnanapragasam JP, et al. Acquired stenosis of normally connected pulmonary veins[J]. Cardiol Young, 2007, 17(3): 322-327. |
| [11] | Drossner DM, Kim DW, Maher KO, et al. Pulmonary vein stenosis: prematurity and associated conditions[J]. Pediatrics, 2008, 122(3): e656-61. PMID: 18762500 |
| [12] | Swier NL, Richards B, Cua CL, et al. Pulmonary vein stenosis in neonates with severe bronchopulmonary dysplasia[J]. Am J Perinatol, 2016, 33(7): 671-677. |
| [13] | Seale AN, Webber S A, Uemura H, et al. Pulmonary vein stenosis: the UK, Ireland and Sweden collaborative study[J]. Heart, 2009, 95(23): 1944-1949. |
| [14] | Vanderlaan RD, Caldarone C A. Pulmonary vein stenosis: incremental knowledge gains to improve outcomes[J]. Children (Basel), 2021, 8(6):481. |
| [15] | Nasr VG, Callahan R, Wichner Z, et al. Intraluminal pulmonary vein stenosis in children: a “ new” lesion[J]. Anesth Analg, 2019, 129(1): 27-40. |
| [16] | Riedlinger WF, Juraszek AL, Jenkins KJ, et al. Pulmonary vein stenosis: expression of receptor tyrosine kinases by lesional cells[J]. Cardiovasc Pathol, 2006, 15(2): 91-99. |
| [17] | Benjamin JT, Hamm CR, Zayek M, et al. Acquired left-sided pulmonary vein stenosis in an extremely premature infant: a new entity?[J]. J Pediatr, 2009, 154(3): 459, |
| [18] | Heching HJ, Turner M, Farkouh-Karoleski C, et al. Pulmonary vein stenosis and necrotising enterocolitis: is there a possible link with necrotising enterocolitis?[J]. Arch Dis Child Fetal Neonatal Ed, 2014, 99(4): F282-285. |
| [19] | Duchon J, Farkouh-Karoleski C, Bailey DD, et al. Association between pulmonary vein stenosis and necrotizing enterocolitis or gastrointestinal pathology: A case-control study[J]. Ann Pediatr Cardiol, 2022, 15(1): 13-19. |
| [20] | DiLorenzo MP, Santo, Rome JJ, et al. Pulmonary vein stenosis: outcomes in children with congenital heart disease and prematurity[J]. Semin Thorac Cardiovasc Surg, 2019, 31(2): 266-273. |
| [21] | Lin Y, Amin EK, Keller RL, et al. Doppler echocardiographic features of pulmonary vein stenosis in ex-preterm children[J]. J Am Soc Echocardiogr, 2022, 35(4): 435-442. |
| [22] | Walters A, Grosse J. Is This My Home? A palliative care journey through life and death in the NICU: a case report[J]. Adv Neonatal Care, 2020, 20(2): 127-135. |
| [23] | Yun TJ, Coles J G, Konstantinov IE, et al. Conventional and sutureless techniques for management of the pulmonary veins: Evolution of indications from postrepair pulmonary vein stenosis to primary pulmonary vein anomalies[J]. J Thorac Cardiovasc Surg, 2005, 129(1): 167-174. |
| [24] | Sokoliuk V, DiNardo J A, Brown ML. Never say never: the use of nitric oxide in patients with obstructed pulmonary veins: a case report[J]. A A Pract, 2019, 12(6): 205-207. |
| [25] | van Duin RWB, Stam K, Uitterdijk A, et al. Intervening with the nitric oxide pathway to alleviate pulmonary hypertension in pulmonary vein stenosis[J]. J Clin Med, 2019, 8(8):1204. |
| [26] | Hilgendorff A., Apitz C., Bonnet D., et al. Pulmonary hypertension associated with acute or chronic lung diseases in the preterm and term neonate and infant. The European Paediatric Pulmonary Vascular Disease Network, endorsed by ISHLT and DGPK[J]. Heart, 2016, 102 Suppl 2: ii49-56. |
| [27] | 夏红萍, 张拥军. 重度支气管肺发育不良的表型特征和治疗策略[J]. 临床儿科杂志, 2022, 40(6): 401-406. |
| [28] | Callahan R, Kieran MW, Baird CW, et al. Adjunct targeted biologic inhibition agents to treat aggressive multivessel intraluminal pediatric pulmonary vein stenosis[J]. J Pediatr, 2018, 198: 29-35 e5. |
| [29] | Zhu J, Ide H, Fu YY, et al. Losartan ameliorates "upstream" pulmonary vein vasculopathy in a piglet model of pulmonary vein stenosis[J]. J Thorac Cardiovasc Surg, 2014, 148(6): 2550-2557. |
| [30] | Patel JD, Briones M, Mandhani M, et al. Systemic sirolimus therapy for infants and children with pulmonary vein stenosis[J]. J Am Coll Cardiol, 2021, 77(22): 2807-2818. |
| [31] | Feng Z, Mao F, Ma K, et al. Clinical outcomes, predictors, and surgical management of primary pulmonary vein stenosis[J]. Ann Thorac Surg, 2022, 113(4): 1239-1247. |
| [32] | Rosenblum JM, Altin HF, Gillespie SE, et al. Management outcomes of primary pulmonary vein stenosis[J]. J Thorac Cardiovasc Surg, 2020, 159(3): 1029-1036. |
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