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本文引用的文献

1
Neonatal Chylothoraces: A 10-Year Experience in a Tertiary Neonatal Referral Centre.新生儿乳糜胸:在一家三级新生儿转诊中心的10年经验
Case Rep Pediatr. 2019 Mar 13;2019:3903598. doi: 10.1155/2019/3903598. eCollection 2019.
2
Propranolol treatment for chylothorax due to diffuse lymphangiomatosis.普萘洛尔治疗弥漫性淋巴管瘤病所致乳糜胸。
Pediatr Blood Cancer. 2019 May;66(5):e27592. doi: 10.1002/pbc.27592. Epub 2019 Jan 4.
3
Octreotide Use in Neonates: A Case Series.奥曲肽在新生儿中的应用:病例系列。
Drugs R D. 2018 Sep;18(3):191-198. doi: 10.1007/s40268-018-0237-9.
4
Sildenafil and Retinopathy of Prematurity in Preterm Infants with Bronchopulmonary Dysplasia.西地那非与支气管肺发育不良早产儿并发早产儿视网膜病变。
J Pediatr. 2018 Aug;199:16-21. doi: 10.1016/j.jpeds.2018.04.005. Epub 2018 May 9.
5
Successful management of extremely high-output refractory congenital chylothorax with chemical pleurodesis using 4% povidone-iodine and propranolol: a case report.使用4%聚维酮碘和普萘洛尔进行化学性胸膜固定术成功治疗极高输出量难治性先天性乳糜胸:一例报告
Clin Case Rep. 2018 Feb 27;6(4):702-708. doi: 10.1002/ccr3.1449. eCollection 2018 Apr.
6
A Case Report of 2 Sirolimus-Related Deaths Among Infants With Kaposiform Hemangioendotheliomas.婴儿 Kaposiform 血管内皮细胞瘤中发生的 2 例与西罗莫司相关的死亡病例报告。
Pediatrics. 2018 Apr;141(Suppl 5):S425-S429. doi: 10.1542/peds.2016-2919.
7
Octreotide for congenital and acquired chylothorax in newborns: A systematic review.奥曲肽用于新生儿先天性和后天性乳糜胸:一项系统评价。
J Paediatr Child Health. 2018 Aug;54(8):840-847. doi: 10.1111/jpc.13889. Epub 2018 Mar 30.
8
Sirolimus for management of complex vascular anomalies - A proposed dosing regimen for very young infants.西罗莫司用于治疗复杂血管异常——一种针对极低龄婴儿的拟用给药方案。
Int J Pediatr Otorhinolaryngol. 2018 Feb;105:48-51. doi: 10.1016/j.ijporl.2017.11.034. Epub 2017 Dec 1.
9
Effects of somatostatin/octreotide treatment in neonates with congenital chylothorax.生长抑素/奥曲肽治疗先天性乳糜胸新生儿的疗效
Medicine (Baltimore). 2017 Jul;96(29):e7594. doi: 10.1097/MD.0000000000007594.
10
Congenital pleural effusions: 15 years single-centre experience from North-East England.先天性胸腔积液:来自英格兰东北部的15年单中心经验。
J Matern Fetal Neonatal Med. 2018 Aug;31(15):2086-2089. doi: 10.1080/14767058.2017.1333097. Epub 2017 Jun 2.

普萘洛尔治疗新生儿淋巴管畸形——病例报告及文献综述

Propranolol for the Treatment of Lymphatic Malformations in a Neonate - A Case Report and Review of Literature.

作者信息

Liviskie Caren J, Brennan Colleen C, McPherson Christopher C, Vesoulis Zachary A

出版信息

J Pediatr Pharmacol Ther. 2020;25(2):155-162. doi: 10.5863/1551-6776-25.2.155.

DOI:10.5863/1551-6776-25.2.155
PMID:32071591
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7025746/
Abstract

Lymphatic malformations in neonates often manifest as a chylothorax, and although rare, morbidity and mortality can be significant. First-line treatment with medium-chain triglyceride-enriched formulas, or enteric rest with total parenteral nutrition, are not always successful. We describe the case of a premature neonate with trisomy 21 who presented with bilateral pleural effusions and a pericardial effusion that worsened with the initiation of enteral nutrition. Clinical improvement was not seen until the initiation of treatment with oral propranolol at a maximum dosage of 0.5 mg/kg/day divided every 8 hours with extubation 8 days after propranolol initiation. Two case reports have described the use of propranolol in similar patients receiving 2 mg/kg/day; however, our experience is the first to report treatment success at a much lower dose. A review of the literature for alternative medication treatments uncovered numerous case reports and series documenting variable results with incongruent definitions of treatment success in a diverse patient population. The rarity of this disease state makes accrual of patients difficult and more robust treatment data unlikely. Therefore, selection of the optimal adjunctive treatment must be based on individual patient and disease state characteristics as well as safety and efficacy profile of the medication.

摘要

新生儿淋巴管畸形常表现为乳糜胸,虽然罕见,但发病率和死亡率可能很高。使用富含中链甘油三酯的配方奶进行一线治疗,或通过全胃肠外营养进行肠道休息,并不总是成功的。我们描述了一例患有21三体综合征的早产儿病例,该患儿出现双侧胸腔积液和心包积液,在开始肠内营养后病情恶化。直到开始使用口服普萘洛尔治疗,最大剂量为0.5mg/kg/天,每8小时一次,在开始使用普萘洛尔8天后拔管,才出现临床改善。有两篇病例报告描述了在类似患者中使用普萘洛尔的剂量为2mg/kg/天;然而,我们的经验是首次报告在低得多的剂量下治疗成功。对替代药物治疗的文献综述发现了大量病例报告和系列研究,记录了不同患者群体中治疗成功定义不一致的情况下的不同结果。这种疾病状态的罕见性使得患者的积累变得困难,更可靠的治疗数据也不太可能获得。因此,选择最佳辅助治疗必须基于个体患者和疾病状态特征以及药物的安全性和有效性。