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Research: Neurodevelopmental Outcomes in Children After Fetoscopic Endoluminal Tracheal Occlusion for Severe Congenital Diaphragmatic Hernia: Results From a Multidisciplinary Clinic

Updated on 3 min read

J Pediatr Surg

. 2024 Mar 20:S0022-3468(24)00195-7.

 doi: 10.1016/j.jpedsurg.2024.03.041. Online ahead of print. https://pubmed.ncbi.nlm.nih.gov/38599907/

Neurodevelopmental Outcomes in Children After Fetoscopic Endoluminal Tracheal Occlusion for Severe Congenital Diaphragmatic Hernia: Results From a Multidisciplinary Clinic

Shelby R Sferra 1, Annalise B Penikis 1, Matthew Guo 1, Ahmet A Baschat 2, Peter J Mogayzel 3, Vera Joanna Burton 4, Shaun M Kunisaki 5

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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.

Conclusion: These initial data suggest that FETO is associated with favorable neurodevelopmental outcomes at 24-months compared to severe CDH under expectant management.

Level of evidence: III.

Keywords: Congenital diaphragmatic hernia; Fetal therapy; Fetal tracheal occlusion; Outcomes.

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