Abstract：
BACKGROUND: We compared early neurodevelopmental morbidity in young children with severe CDH who underwent FETO to those without fetal therapy.
METHODS: We conducted a prospective study of severe CDH patients undergoing FETO (n = 18) at a single North American center from 2015 to 2021 (NCT02710968). Outpatient survivors (n = 12) were evaluated by a multidisciplinary team and compared to expectantly managed CDH patients. Neurodevelopmental outcomes were assessed using the Capute Scales [Clinical Linguistic and Auditory Milestone Scales (CLAMS) and Cognitive Adaptive Test (CAT)], with a developmental quotient (DQ) < 85 indicative of at-risk for delay.
RESULTS: At one year, 58% (n = 7) of FETO patients underwent evaluation, with notable concern for language delay (CLAMS median DQ, 80.1 [interquartile range, 67.6-86.7]). FETO scores improved by 24-months, whereas high severity/non-FETO scores declined [CLAMS median DQ (Difference in DQ), 92.3 (+12.2) vs. 77.1 (-13.4), respectively; p = 0.049]. On the initial CAT, FETO patients had concern for visual motor and problem-solving delays, with a median DQ of 81.3 (62.1-89.4). At 24-months, FETO patients had improving scores [Median CAT DQ, 90.8 (+9.5)], whereas high severity/non-FETO [87.5 (-3.0), p = 0.28] had declining scores.
CONCLUSIONS: These initial data suggest that FETO is associated with favorable neurodevelopmental outcomes at 24-months compared to severe CDH under expectant management.
METHODS: III.
METHODS: We conducted a prospective study of severe CDH patients undergoing FETO (n = 18) at a single North American center from 2015 to 2021 (NCT02710968). Outpatient survivors (n = 12) were evaluated by a multidisciplinary team and compared to expectantly managed CDH patients. Neurodevelopmental outcomes were assessed using the Capute Scales [Clinical Linguistic and Auditory Milestone Scales (CLAMS) and Cognitive Adaptive Test (CAT)], with a developmental quotient (DQ) < 85 indicative of at-risk for delay.
RESULTS: At one year, 58% (n = 7) of FETO patients underwent evaluation, with notable concern for language delay (CLAMS median DQ, 80.1 [interquartile range, 67.6-86.7]). FETO scores improved by 24-months, whereas high severity/non-FETO scores declined [CLAMS median DQ (Difference in DQ), 92.3 (+12.2) vs. 77.1 (-13.4), respectively; p = 0.049]. On the initial CAT, FETO patients had concern for visual motor and problem-solving delays, with a median DQ of 81.3 (62.1-89.4). At 24-months, FETO patients had improving scores [Median CAT DQ, 90.8 (+9.5)], whereas high severity/non-FETO [87.5 (-3.0), p = 0.28] had declining scores.
CONCLUSIONS: These initial data suggest that FETO is associated with favorable neurodevelopmental outcomes at 24-months compared to severe CDH under expectant management.
METHODS: III.
摘要：
背景：我们比较了接受FETO治疗的重度CDH患儿与未接受胎儿治疗的患儿的早期神经发育发病率。
方法：我们对2015年至2021年在北美一家中心接受FETO（n=18）的重度CDH患者进行了一项前瞻性研究（NCT02710968）。门诊幸存者（n=12）由多学科小组进行评估，并与预期管理的CDH患者进行比较。使用Capute量表[临床语言和听觉里程碑量表（CLAMS）和认知适应测验（CAT）]评估神经发育结果。发育商（DQ）<85表明有延迟的风险。
结果：一年后，58%(n=7)的FETO患者接受了评估，值得注意的是对语言延迟的关注(CLAMS中值DQ，80.1[四分位数间距，67.6-86.7]).FETO分数提高了24个月，而高严重程度/非FETO评分下降[CLAMS中位数DQ（DQ差异），92.3（+12.2）vs.77.1(-13.4),分别为；p=0.049]。在最初的CAT上，FETO患者担心视觉运动和解决问题的延迟，中位数DQ为81.3（62.1-89.4）。24个月时,FETO患者的评分有所改善[CATDQ中位数，90.8(+9.5)],而高严重性/非FETO[87.5（-3.0），p=0.28]得分下降。
结论：这些初步数据表明，与预期管理下的重度CDH相比，FETO在24个月时与良好的神经发育结局相关。
方法：III.
方法：我们对2015年至2021年在北美一家中心接受FETO（n=18）的重度CDH患者进行了一项前瞻性研究（NCT02710968）。门诊幸存者（n=12）由多学科小组进行评估，并与预期管理的CDH患者进行比较。使用Capute量表[临床语言和听觉里程碑量表（CLAMS）和认知适应测验（CAT）]评估神经发育结果。发育商（DQ）<85表明有延迟的风险。
结果：一年后，58%(n=7)的FETO患者接受了评估，值得注意的是对语言延迟的关注(CLAMS中值DQ，80.1[四分位数间距，67.6-86.7]).FETO分数提高了24个月，而高严重程度/非FETO评分下降[CLAMS中位数DQ（DQ差异），92.3（+12.2）vs.77.1(-13.4),分别为；p=0.049]。在最初的CAT上，FETO患者担心视觉运动和解决问题的延迟，中位数DQ为81.3（62.1-89.4）。24个月时,FETO患者的评分有所改善[CATDQ中位数，90.8(+9.5)],而高严重性/非FETO[87.5（-3.0），p=0.28]得分下降。
结论：这些初步数据表明，与预期管理下的重度CDH相比，FETO在24个月时与良好的神经发育结局相关。
方法：III.
