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Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?

Previous studies have frequently reported neurocognitive deficits in children born with congenital diaphragmatic hernia (CDH) at school age, which may contribute to academic difficulties. Yet, age at onset of these deficits is currently unknown. We evaluated neurocognitive skills with possible deter...

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Autores principales: de Munck, Sophie, Otter, Suzan C. M. Cochius-den, Schnater, J. Marco, van Rosmalen, Joost, Peters, Nina C. J., van Gils-Frijters, Annabel P. J. M., van Haren, Neeltje E. M., Gischler, Saskia J., IJsselstijn, Hanneke, Rietman, André B.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10023636/
https://www.ncbi.nlm.nih.gov/pubmed/36565322
http://dx.doi.org/10.1007/s00431-022-04696-1
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author de Munck, Sophie
Otter, Suzan C. M. Cochius-den
Schnater, J. Marco
van Rosmalen, Joost
Peters, Nina C. J.
van Gils-Frijters, Annabel P. J. M.
van Haren, Neeltje E. M.
Gischler, Saskia J.
IJsselstijn, Hanneke
Rietman, André B.
author_facet de Munck, Sophie
Otter, Suzan C. M. Cochius-den
Schnater, J. Marco
van Rosmalen, Joost
Peters, Nina C. J.
van Gils-Frijters, Annabel P. J. M.
van Haren, Neeltje E. M.
Gischler, Saskia J.
IJsselstijn, Hanneke
Rietman, André B.
author_sort de Munck, Sophie
collection PubMed
description Previous studies have frequently reported neurocognitive deficits in children born with congenital diaphragmatic hernia (CDH) at school age, which may contribute to academic difficulties. Yet, age at onset of these deficits is currently unknown. We evaluated neurocognitive skills with possible determinants in preschool children born with CDH. Eligible 5-year-old children born with CDH (2010–2015) who participated in our prospective structural follow-up program were included. We used the WPPSI-III to assess intelligence, subtests of the Kaufman-ABC for memory, and NEPSY-II to assess inhibition and attention. We included 63 children. Their test scores generally were within or significantly above normal range: total IQ = 103.4 (15.7) (p = 0.13); Verbal memory = 10.2 (2.8) (p = 0.61); Visuospatial memory = 11.4 (2.6) (p < 0.01); Inhibition = 10.5 (2.2), (p = 0.10). In univariable analyses, length of ICU-stay was negatively associated with IQ, and maximum vasoactive inotropic score and open repair were negatively associated with inhibition skills. In multivariable regression analysis, the latter association remained (B = 5.52, p = 0.04 (CI 0.32–10.72)).    Conclusions: In these tested 5-year-old children born with CDH, neuropsychological outcome was normal on average. While problems in 8-year-olds are common, we did not detect onset of these problems at age 5. Yet, we cannot rule out that this cohort had a relatively mild level of disease severity; therefore, conclusions should be interpreted with caution. However, given the growing-into-deficit hypothesis, meaning that deviant brain development in early life is revealed once higher cognitive brain functions are demanded, follow-up should be conducted up to school age, and preferably beyond. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00431-022-04696-1.
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spelling pubmed-100236362023-03-19 Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell? de Munck, Sophie Otter, Suzan C. M. Cochius-den Schnater, J. Marco van Rosmalen, Joost Peters, Nina C. J. van Gils-Frijters, Annabel P. J. M. van Haren, Neeltje E. M. Gischler, Saskia J. IJsselstijn, Hanneke Rietman, André B. Eur J Pediatr Research Previous studies have frequently reported neurocognitive deficits in children born with congenital diaphragmatic hernia (CDH) at school age, which may contribute to academic difficulties. Yet, age at onset of these deficits is currently unknown. We evaluated neurocognitive skills with possible determinants in preschool children born with CDH. Eligible 5-year-old children born with CDH (2010–2015) who participated in our prospective structural follow-up program were included. We used the WPPSI-III to assess intelligence, subtests of the Kaufman-ABC for memory, and NEPSY-II to assess inhibition and attention. We included 63 children. Their test scores generally were within or significantly above normal range: total IQ = 103.4 (15.7) (p = 0.13); Verbal memory = 10.2 (2.8) (p = 0.61); Visuospatial memory = 11.4 (2.6) (p < 0.01); Inhibition = 10.5 (2.2), (p = 0.10). In univariable analyses, length of ICU-stay was negatively associated with IQ, and maximum vasoactive inotropic score and open repair were negatively associated with inhibition skills. In multivariable regression analysis, the latter association remained (B = 5.52, p = 0.04 (CI 0.32–10.72)).    Conclusions: In these tested 5-year-old children born with CDH, neuropsychological outcome was normal on average. While problems in 8-year-olds are common, we did not detect onset of these problems at age 5. Yet, we cannot rule out that this cohort had a relatively mild level of disease severity; therefore, conclusions should be interpreted with caution. However, given the growing-into-deficit hypothesis, meaning that deviant brain development in early life is revealed once higher cognitive brain functions are demanded, follow-up should be conducted up to school age, and preferably beyond. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00431-022-04696-1. Springer Berlin Heidelberg 2022-12-24 2023 /pmc/articles/PMC10023636/ /pubmed/36565322 http://dx.doi.org/10.1007/s00431-022-04696-1 Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Research
de Munck, Sophie
Otter, Suzan C. M. Cochius-den
Schnater, J. Marco
van Rosmalen, Joost
Peters, Nina C. J.
van Gils-Frijters, Annabel P. J. M.
van Haren, Neeltje E. M.
Gischler, Saskia J.
IJsselstijn, Hanneke
Rietman, André B.
Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
title Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
title_full Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
title_fullStr Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
title_full_unstemmed Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
title_short Neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
title_sort neuropsychological outcome in survivors of congenital diaphragmatic hernia at 5 years of age, what does it tell?
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10023636/
https://www.ncbi.nlm.nih.gov/pubmed/36565322
http://dx.doi.org/10.1007/s00431-022-04696-1
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