Abstract：
BACKGROUND: Temporary fetoscopic endoluminal tracheal occlusion (FETO) promotes lung growth and increases survival in selected fetuses with congenital diaphragmatic hernia (CDH). FETO is performed percutaneously by inserting into the trachea a balloon designed for vascular occlusion. However, reports on the potential postnatal side-effects of the balloon are scarce. This study aimed to evaluate the prevalence of tracheomalacia in infants with CDH managed with and without FETO and other consequences related to the use of the balloon.
METHODS: In this multicentre, retrospective cohort study, we included infants who were live born with CDH, either with FETO or without, who were managed postnatally at four centres (UZ Leuven, Leuven, Belgium; Antoine Béclère, Clamart, France; BCNatal, Barcelona, Spain; and HCor-Heart Hospital, São Paulo, Brazil) between April 5, 2002, and June 2, 2021. We primarily assessed the prevalence of all (symptomatic and asymptomatic) tracheomalacia as reported in medical records among infants with and without FETO. Secondarily we assessed the prevalence of symptomatic tracheomalacia and its resolution as reported in medical records, and compared tracheal diameters as measured on postnatal x-rays. Crude and adjusted risk ratios (aRRs) and 95% CIs were calculated via modified Poisson regression models with robust error variances for potential association between FETO and tracheomalacia. Variables included in the adjusted model were the side of the hernia, observed-to-expected lung-to-head ratio, and gestational age at birth. Crude and adjusted mean differences and 95% CIs were calculated via linear regression models to assess the presence and magnitude of association between FETO and tracheal diameters. In infants who had undergone FETO we also assessed the localisation of balloon remnants on x-rays, and the methods used for reversal of occlusion and potential complications associated with balloon remnants as documented in clinical records. Finally we investigated whether the presence of balloon remnants was influenced by the interval between balloon removal and delivery.
RESULTS: 505 neonates were included in the study, of whom 287 had undergone FETO and 218 had not. Tracheomalacia was reported in 18 (6%) infants who had undergone FETO and in three (1%) who had not (aRR 6·17 [95% CI 1·83-20·75]; p=0·0030). Tracheomalacia was first reported in the FETO group at a median of 5·0 months (IQR 0·8-13·0). Symptomatic tracheomalacia was reported in 13 (5%) infants who had undergone FETO, which resolved in ten (77%) children by 55·0 months (IQR 14·0-83·0). On average, infants who had undergone FETO had a 31·3% wider trachea (with FETO tracheal diameter 7·43 mm [SD 1·24], without FETO tracheal diameter 5·10 mm [SD 0·84]; crude mean difference 2·32 [95% CI 2·11-2·54]; p<0·0001; adjusted mean difference 2·62 [95% CI 2·35-2·89]; p<0·0001). At birth, the metallic component was visible within the body in 75 (37%) of 205 infants with available thoraco-abdominal x-rays: it was located in the gastrointestinal tract in 60 (80%) and in the lung in 15 (20%). No side-effects were reported for any of the infants during follow-up. The metallic component was more likely to be in the lung than either outside the body or the gastrointestinal tract when the interval between occlusion reversal and birth was less than 24 h.
CONCLUSIONS: Although FETO was associated with an increased tracheal diameter and an increased probability of tracheomalacia, symptomatic tracheomalacia typically resolved over time. There is a higher risk of retention of metallic balloon components if reversal of the occlusion occurs less than 24 h before delivery. Finally, there were no reported side-effects of the metallic component of the balloon persisting in the body during follow-up. Longer-term follow-up is needed to ensure that no tracheal problems arise later in life.
BACKGROUND: None.
METHODS: In this multicentre, retrospective cohort study, we included infants who were live born with CDH, either with FETO or without, who were managed postnatally at four centres (UZ Leuven, Leuven, Belgium; Antoine Béclère, Clamart, France; BCNatal, Barcelona, Spain; and HCor-Heart Hospital, São Paulo, Brazil) between April 5, 2002, and June 2, 2021. We primarily assessed the prevalence of all (symptomatic and asymptomatic) tracheomalacia as reported in medical records among infants with and without FETO. Secondarily we assessed the prevalence of symptomatic tracheomalacia and its resolution as reported in medical records, and compared tracheal diameters as measured on postnatal x-rays. Crude and adjusted risk ratios (aRRs) and 95% CIs were calculated via modified Poisson regression models with robust error variances for potential association between FETO and tracheomalacia. Variables included in the adjusted model were the side of the hernia, observed-to-expected lung-to-head ratio, and gestational age at birth. Crude and adjusted mean differences and 95% CIs were calculated via linear regression models to assess the presence and magnitude of association between FETO and tracheal diameters. In infants who had undergone FETO we also assessed the localisation of balloon remnants on x-rays, and the methods used for reversal of occlusion and potential complications associated with balloon remnants as documented in clinical records. Finally we investigated whether the presence of balloon remnants was influenced by the interval between balloon removal and delivery.
RESULTS: 505 neonates were included in the study, of whom 287 had undergone FETO and 218 had not. Tracheomalacia was reported in 18 (6%) infants who had undergone FETO and in three (1%) who had not (aRR 6·17 [95% CI 1·83-20·75]; p=0·0030). Tracheomalacia was first reported in the FETO group at a median of 5·0 months (IQR 0·8-13·0). Symptomatic tracheomalacia was reported in 13 (5%) infants who had undergone FETO, which resolved in ten (77%) children by 55·0 months (IQR 14·0-83·0). On average, infants who had undergone FETO had a 31·3% wider trachea (with FETO tracheal diameter 7·43 mm [SD 1·24], without FETO tracheal diameter 5·10 mm [SD 0·84]; crude mean difference 2·32 [95% CI 2·11-2·54]; p<0·0001; adjusted mean difference 2·62 [95% CI 2·35-2·89]; p<0·0001). At birth, the metallic component was visible within the body in 75 (37%) of 205 infants with available thoraco-abdominal x-rays: it was located in the gastrointestinal tract in 60 (80%) and in the lung in 15 (20%). No side-effects were reported for any of the infants during follow-up. The metallic component was more likely to be in the lung than either outside the body or the gastrointestinal tract when the interval between occlusion reversal and birth was less than 24 h.
CONCLUSIONS: Although FETO was associated with an increased tracheal diameter and an increased probability of tracheomalacia, symptomatic tracheomalacia typically resolved over time. There is a higher risk of retention of metallic balloon components if reversal of the occlusion occurs less than 24 h before delivery. Finally, there were no reported side-effects of the metallic component of the balloon persisting in the body during follow-up. Longer-term follow-up is needed to ensure that no tracheal problems arise later in life.
BACKGROUND: None.
摘要：
背景：在某些患有先天性膈疝（CDH）的胎儿中，临时胎儿镜下腔内气管阻塞（FETO）可促进肺生长并增加存活率。通过将设计用于血管闭塞的球囊插入气管中来经皮执行FETO。然而,关于气球潜在的产后副作用的报道很少。这项研究旨在评估有或没有FETO的CDH婴儿气管软化的患病率，以及与使用球囊相关的其他后果。
方法：在这个多中心中，回顾性队列研究，我们包括患有CDH的婴儿，无论有或没有，谁在出生后在四个中心进行管理(UZLeuven，鲁汶,比利时；安托万·贝克莱，克拉马特,法国；BCNatal,巴塞罗那,西班牙；和Hcor心脏医院，圣保罗,巴西)在2002年4月5日至2021年6月2日之间。我们主要评估了所有（有症状和无症状）气管软化的患病率，如医疗记录中报告的有和无FETO的婴儿。其次，我们评估了病历中报告的症状性气管软化的患病率及其解决方法，并比较了出生后X射线测得的气管直径。通过改进的Poisson回归模型计算粗风险比和调整后的风险比（aRR）和95％CIs，该模型具有可靠的误差方差，可用于FETO和气管软化之间的潜在关联。调整后的模型中包含的变量是疝气的侧面，观察到的预期肺头比，和出生时的胎龄。通过线性回归模型计算粗和调整后的平均差异和95％CIs，以评估FETO和气管直径之间的关联的存在和程度。在接受过FETO的婴儿中，我们还评估了X射线上气球残留物的定位，以及临床记录中记录的用于逆转与球囊残留相关的闭塞和潜在并发症的方法。最后，我们调查了球囊残留物的存在是否受到球囊移除和递送之间的间隔的影响。
结果：505例新生儿被纳入研究，其中287人接受了FETO，218人没有。据报道，有18例（6％）接受FETO的婴儿和3例（1％）未接受FETO的婴儿发生气管软化（aRR6·17[95％CI1·83-20·75]；p=0·0030）。FETO组首次报告气管软化的中位数为5·0个月（IQR0·8-13·0）。有症状的气管软化在13（5％）婴儿谁经历了FETO报告，到了55·0个月（IQR14·0-83·0），10名（77%）儿童得以解决。平均而言,接受FETO的婴儿气管宽31·3％（FETO气管直径7·43mm[SD1·24]，无FETO气管直径5·10mm[SD0·84]；粗平均差2·32[95%CI2·11-2·54]；p<0·0001；调整平均差2·62[95%CI2·35-2·89]；p<0·0001）。出生时,在205例具有胸腹X线的婴儿中，有75例（37％）在体内可见金属成分：60例（80％）位于胃肠道，15例（20％）位于肺部。随访期间未报告任何婴儿的副作用。当闭塞逆转与出生之间的间隔小于24小时时，金属成分更有可能在肺中，而不是在体外或胃肠道中。
结论：尽管FETO与气管直径增加和气管软化的可能性增加有关，症状性气管软化通常随时间缓解.如果在分娩前24小时内发生闭塞逆转，则金属球囊组件的滞留风险更高。最后,在随访期间,球囊的金属成分未出现持续存在于体内的副作用.需要长期随访，以确保以后的生活中不会出现气管问题。
背景：无。
方法：在这个多中心中，回顾性队列研究，我们包括患有CDH的婴儿，无论有或没有，谁在出生后在四个中心进行管理(UZLeuven，鲁汶,比利时；安托万·贝克莱，克拉马特,法国；BCNatal,巴塞罗那,西班牙；和Hcor心脏医院，圣保罗,巴西)在2002年4月5日至2021年6月2日之间。我们主要评估了所有（有症状和无症状）气管软化的患病率，如医疗记录中报告的有和无FETO的婴儿。其次，我们评估了病历中报告的症状性气管软化的患病率及其解决方法，并比较了出生后X射线测得的气管直径。通过改进的Poisson回归模型计算粗风险比和调整后的风险比（aRR）和95％CIs，该模型具有可靠的误差方差，可用于FETO和气管软化之间的潜在关联。调整后的模型中包含的变量是疝气的侧面，观察到的预期肺头比，和出生时的胎龄。通过线性回归模型计算粗和调整后的平均差异和95％CIs，以评估FETO和气管直径之间的关联的存在和程度。在接受过FETO的婴儿中，我们还评估了X射线上气球残留物的定位，以及临床记录中记录的用于逆转与球囊残留相关的闭塞和潜在并发症的方法。最后，我们调查了球囊残留物的存在是否受到球囊移除和递送之间的间隔的影响。
结果：505例新生儿被纳入研究，其中287人接受了FETO，218人没有。据报道，有18例（6％）接受FETO的婴儿和3例（1％）未接受FETO的婴儿发生气管软化（aRR6·17[95％CI1·83-20·75]；p=0·0030）。FETO组首次报告气管软化的中位数为5·0个月（IQR0·8-13·0）。有症状的气管软化在13（5％）婴儿谁经历了FETO报告，到了55·0个月（IQR14·0-83·0），10名（77%）儿童得以解决。平均而言,接受FETO的婴儿气管宽31·3％（FETO气管直径7·43mm[SD1·24]，无FETO气管直径5·10mm[SD0·84]；粗平均差2·32[95%CI2·11-2·54]；p<0·0001；调整平均差2·62[95%CI2·35-2·89]；p<0·0001）。出生时,在205例具有胸腹X线的婴儿中，有75例（37％）在体内可见金属成分：60例（80％）位于胃肠道，15例（20％）位于肺部。随访期间未报告任何婴儿的副作用。当闭塞逆转与出生之间的间隔小于24小时时，金属成分更有可能在肺中，而不是在体外或胃肠道中。
结论：尽管FETO与气管直径增加和气管软化的可能性增加有关，症状性气管软化通常随时间缓解.如果在分娩前24小时内发生闭塞逆转，则金属球囊组件的滞留风险更高。最后,在随访期间,球囊的金属成分未出现持续存在于体内的副作用.需要长期随访，以确保以后的生活中不会出现气管问题。
背景：无。
