新生儿胎粪吸入综合征与ABCA3基因突变和支原体感染:1例报告。

IF 2 3区 医学 Q2 PEDIATRICS
Oliver Stelzig, Beatrix Mühlegger, Anna Zschocke, Ursula Kiechl-Kohlendorfer, Elke Griesmaier
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引用次数: 0

摘要

早产儿发生呼吸窘迫综合征(RDS)的风险很高。编码表面活性剂蛋白B和C或atp结合盒转运蛋白A3 (ABCA3)的基因突变是罕见的,但已知与严重的RDS和间质性肺疾病有关。这些突变在一般人群中的确切流行率很难确定,因为它们通常与临床症状有关。由于表达或诊断的差异,大多数病例未被发现。据估计,它们影响一小部分人口,ABCA3突变最常被发现与新生儿的严重肺部疾病有关。即使abca3杂合突变也会增加新生儿RDS的风险和严重程度。这些蛋白的表达受发育调控,随胎龄增加,对出生时肺表面活性物质的功能至关重要。其他肺部压力源,如胎粪吸入综合征或肺部感染,可导致与严重病程相关的复杂临床症状。本病例报告描述了一例极早产女婴,疑似胎粪吸入综合征,严重RDS,肺炎支原体感染,abca3杂合突变。该报告讨论了临床表现、诊断评估和治疗干预措施,强调了极端早产背景下多发性肺部疾病的复杂性。在生存能力的极限下,与年龄较大的儿童相比,严重呼吸功能不全的治疗选择有限。长期相对缺氧后的神经发育预后是讨论改变治疗目标时需要考虑的关键因素。特别是在严重的情况下,必须考虑肺部感染和表面活性物质代谢的遗传变化。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Neonatal meconium aspiration syndrome associated with ABCA3 gene mutation and mycoplasma infection: a case report.

Preterm infants are at high risk of developing respiratory distress syndrome (RDS). Mutations in the genes encoding for surfactant proteins B and C or the ATP-binding cassette transporter A3 (ABCA3) are rare but known to be associated with severe RDS and interstitial lung diseases. The exact prevalence of these mutations in the general population is difficult to determine, as they are usually studied in connection with clinical symptoms. Most cases are not captured due to variability in expression or diagnosis. It is estimated that they affect a small percentage of the population, with mutations in ABCA3 most commonly identified in association with severe lung diseases in newborns. Even heterozygous ABCA3-mutations can increase the risk and severity of RDS in neonates. The expression of these proteins is developmentally regulated, increases with gestational age, and is crucial for the function of pulmonary surfactant at birth. Additional lung stressors, such as meconium aspiration syndrome or pulmonary infections, can lead to a complex clinical picture associated with severe courses. This case report describes an extremely preterm female infant with suspected meconium aspiration syndrome, severe RDS, Mycoplasma pneumoniae infection, and a heterozygous ABCA3-mutation. The report discusses the clinical presentation, diagnostic evaluation, and therapeutic interventions, emphasizing the complexities associated with multiple pulmonary conditions in the context of extreme prematurity. At the limits of viability, therapeutic options for severe respiratory insufficiency are limited compared to older children. The developmental neurological prognosis following prolonged relative hypoxia is a crucial factor to consider in discussions about changing treatment goals. Particularly in severe cases, pulmonary infections and genetic changes in surfactant metabolism must be considered in newborns with RDS.

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来源期刊
BMC Pediatrics
BMC Pediatrics PEDIATRICS-
CiteScore
3.70
自引率
4.20%
发文量
683
审稿时长
3-8 weeks
期刊介绍: BMC Pediatrics is an open access journal publishing peer-reviewed research articles in all aspects of health care in neonates, children and adolescents, as well as related molecular genetics, pathophysiology, and epidemiology.
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