Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion

D. Emam; M. Aertsen; L. Van der Veeken; L. Fidon; P. Patkee; V. Kyriakopoulou; L. De Catte; F. Russo; P. Demaerel; T. Vercauteren; M. Rutherford; J. Deprest

doi:10.3174/ajnr.A7760

Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion

D. Emam, M. Aertsen, L. Van der Veeken, L. Fidon, P. Patkee, V. Kyriakopoulou, L. De Catte, F. Russo, P. Demaerel, T. Vercauteren, M. Rutherford, J. Deprest

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Abstract

BACKGROUND AND PURPOSE: Congenital diaphragmatic hernia is associated with high mortality and morbidity, including evidence suggesting neurodevelopmental comorbidities after birth. The aim of this study was to document longitudinal changes in brain biometry and the cortical folding pattern in fetuses with congenital diaphragmatic hernia compared with healthy fetuses. MATERIALS AND METHODS: This is a retrospective cohort study including fetuses with isolated congenital diaphragmatic hernia between January 2007 and May 2019, with at least 2 MR imaging examinations. For controls, we used images from fetuses who underwent MR imaging for an unrelated condition that did not compromise fetal brain development and fetuses from healthy pregnant women. Biometric measurements and 3D segmentations of brain structures were used as well as qualitative and quantitative grading of the supratentorial brain. Brain development was correlated with disease-severity markers. RESULTS: Forty-two fetuses were included, with a mean gestational age at first MR imaging of 28.0 (SD, 2.1) weeks and 33.2 (SD, 1.3) weeks at the second imaging. The mean gestational age in controls was 30.7 (SD, 4.2) weeks. At 28 weeks, fetuses with congenital diaphragmatic hernia had abnormal qualitative and quantitative maturation, more extra-axial fluid, and larger total skull volume. By 33 weeks, qualitative grading scores were still abnormal, but quantitative scoring was in the normal range. In contrast, the extra-axial fluid volume remained abnormal with increased ventricular volume. Normal brain parenchymal volumes were found. CONCLUSIONS: Brain development in fetuses with congenital diaphragmatic hernia around 28 weeks appears to be delayed. This feature is less prominent at 33 weeks. At this stage, there was also an increase in ventricular and extra-axial space volume.

Original language	English
Pages (from-to)	205-211
Number of pages	7
Journal	AJNR. American journal of neuroradiology
Volume	44
Issue number	2
DOIs	https://doi.org/10.3174/ajnr.A7760
Publication status	Published - 1 Feb 2023

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Emam, D., Aertsen, M., Van der Veeken, L., Fidon, L., Patkee, P., Kyriakopoulou, V., De Catte, L., Russo, F., Demaerel, P., Vercauteren, T., Rutherford, M., & Deprest, J. (2023). Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion. AJNR. American journal of neuroradiology, 44(2), 205-211. https://doi.org/10.3174/ajnr.A7760

@article{1bbeac7a35744526952277634727d7a6,

title = "Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion",

abstract = "BACKGROUND AND PURPOSE: Congenital diaphragmatic hernia is associated with high mortality and morbidity, including evidence suggesting neurodevelopmental comorbidities after birth. The aim of this study was to document longitudinal changes in brain biometry and the cortical folding pattern in fetuses with congenital diaphragmatic hernia compared with healthy fetuses. MATERIALS AND METHODS: This is a retrospective cohort study including fetuses with isolated congenital diaphragmatic hernia between January 2007 and May 2019, with at least 2 MR imaging examinations. For controls, we used images from fetuses who underwent MR imaging for an unrelated condition that did not compromise fetal brain development and fetuses from healthy pregnant women. Biometric measurements and 3D segmentations of brain structures were used as well as qualitative and quantitative grading of the supratentorial brain. Brain development was correlated with disease-severity markers. RESULTS: Forty-two fetuses were included, with a mean gestational age at first MR imaging of 28.0 (SD, 2.1) weeks and 33.2 (SD, 1.3) weeks at the second imaging. The mean gestational age in controls was 30.7 (SD, 4.2) weeks. At 28 weeks, fetuses with congenital diaphragmatic hernia had abnormal qualitative and quantitative maturation, more extra-axial fluid, and larger total skull volume. By 33 weeks, qualitative grading scores were still abnormal, but quantitative scoring was in the normal range. In contrast, the extra-axial fluid volume remained abnormal with increased ventricular volume. Normal brain parenchymal volumes were found. CONCLUSIONS: Brain development in fetuses with congenital diaphragmatic hernia around 28 weeks appears to be delayed. This feature is less prominent at 33 weeks. At this stage, there was also an increase in ventricular and extra-axial space volume.",

author = "D. Emam and M. Aertsen and {Van der Veeken}, L. and L. Fidon and P. Patkee and V. Kyriakopoulou and {De Catte}, L. and F. Russo and P. Demaerel and T. Vercauteren and M. Rutherford and J. Deprest",

note = "Funding Information: Received April 28, 2022; accepted after revision December 10. From the Department of Development and Regeneration (D.E., L.V.d.V., L.D.C., F.R., J.D.), Cluster Woman and Child, Group Biomedical Sciences, KU Leuven University of Leuven, Leuven, Belgium; Department Obstetrics and Gynaecology (D.E., L.F.), Faculty of Medicine, Tanta University, Tanta, Egypt; Department of Imaging and Pathology (M.A., P.D.), Clinical Department of Radiology, University Hospitals, KU Leuven, Leuven, Belgium; Clinical Department Obstetrics and Gynaecology (L.V.d.V., L.D.C., F.R., J.D.), University Hospitals Leuven, Leuven, Belgium; Centre for the Developing Brain (P.P., V.K., M.R., J.D.), Division of Imaging Sciences and Biomedical Engineering, Perinatal Imaging and Health and School of Biomedical Engineering and Imaging Sciences (L.F., T.V., J.D.), King{\textquoteright}s College London, King{\textquoteright}s Health Partners, St. Thomas{\textquoteright} Hospital, London, UK; and Institute for Women{\textquoteright}s Health (J.D.), University College London, London, UK. D. Emam and M. Aertsen contributed equally to this work. D.E. is funded by the Egyptian Educational Scholarship from the Ministry of Higher Education and Scientific Research, Egypt. L.V.d.V. is funded by the Erasmus1 Program of the European Union (framework agreement no. 2013-0040). J.D. and T.V. receive support from the Wellcome Trust (WT101957) and the Engineering and Physical Sciences Research Council (NS/A000027/1). J.D. receives support from the Great Ormond Street Hospital Charity Fund. To support L.F. and T.V., this project has received funding from the European Union{\textquoteright}s Horizon 2020 research and innovation program under the Marie Sklodowska-Curie grant agreement No 765148. Publisher Copyright: {\textcopyright} 2023 American Society of Neuroradiology. All rights reserved.",

year = "2023",

month = feb,

day = "1",

doi = "10.3174/ajnr.A7760",

language = "English",

volume = "44",

pages = "205--211",

journal = "AJNR. American journal of neuroradiology",

issn = "0195-6108",

publisher = "American Society of Neuroradiology",

number = "2",

}

Emam, D, Aertsen, M, Van der Veeken, L, Fidon, L , Patkee, P , Kyriakopoulou, V, De Catte, L, Russo, F, Demaerel, P, Vercauteren, T , Rutherford, M & Deprest, J 2023, 'Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion', AJNR. American journal of neuroradiology, vol. 44, no. 2, pp. 205-211. https://doi.org/10.3174/ajnr.A7760

TY - JOUR

T1 - Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion

AU - Emam, D.

AU - Aertsen, M.

AU - Van der Veeken, L.

AU - Fidon, L.

AU - Patkee, P.

AU - Kyriakopoulou, V.

AU - De Catte, L.

AU - Russo, F.

AU - Demaerel, P.

AU - Vercauteren, T.

AU - Rutherford, M.

AU - Deprest, J.

N1 - Funding Information: Received April 28, 2022; accepted after revision December 10. From the Department of Development and Regeneration (D.E., L.V.d.V., L.D.C., F.R., J.D.), Cluster Woman and Child, Group Biomedical Sciences, KU Leuven University of Leuven, Leuven, Belgium; Department Obstetrics and Gynaecology (D.E., L.F.), Faculty of Medicine, Tanta University, Tanta, Egypt; Department of Imaging and Pathology (M.A., P.D.), Clinical Department of Radiology, University Hospitals, KU Leuven, Leuven, Belgium; Clinical Department Obstetrics and Gynaecology (L.V.d.V., L.D.C., F.R., J.D.), University Hospitals Leuven, Leuven, Belgium; Centre for the Developing Brain (P.P., V.K., M.R., J.D.), Division of Imaging Sciences and Biomedical Engineering, Perinatal Imaging and Health and School of Biomedical Engineering and Imaging Sciences (L.F., T.V., J.D.), King’s College London, King’s Health Partners, St. Thomas’ Hospital, London, UK; and Institute for Women’s Health (J.D.), University College London, London, UK. D. Emam and M. Aertsen contributed equally to this work. D.E. is funded by the Egyptian Educational Scholarship from the Ministry of Higher Education and Scientific Research, Egypt. L.V.d.V. is funded by the Erasmus1 Program of the European Union (framework agreement no. 2013-0040). J.D. and T.V. receive support from the Wellcome Trust (WT101957) and the Engineering and Physical Sciences Research Council (NS/A000027/1). J.D. receives support from the Great Ormond Street Hospital Charity Fund. To support L.F. and T.V., this project has received funding from the European Union’s Horizon 2020 research and innovation program under the Marie Sklodowska-Curie grant agreement No 765148. Publisher Copyright: © 2023 American Society of Neuroradiology. All rights reserved.

PY - 2023/2/1

Y1 - 2023/2/1

N2 - BACKGROUND AND PURPOSE: Congenital diaphragmatic hernia is associated with high mortality and morbidity, including evidence suggesting neurodevelopmental comorbidities after birth. The aim of this study was to document longitudinal changes in brain biometry and the cortical folding pattern in fetuses with congenital diaphragmatic hernia compared with healthy fetuses. MATERIALS AND METHODS: This is a retrospective cohort study including fetuses with isolated congenital diaphragmatic hernia between January 2007 and May 2019, with at least 2 MR imaging examinations. For controls, we used images from fetuses who underwent MR imaging for an unrelated condition that did not compromise fetal brain development and fetuses from healthy pregnant women. Biometric measurements and 3D segmentations of brain structures were used as well as qualitative and quantitative grading of the supratentorial brain. Brain development was correlated with disease-severity markers. RESULTS: Forty-two fetuses were included, with a mean gestational age at first MR imaging of 28.0 (SD, 2.1) weeks and 33.2 (SD, 1.3) weeks at the second imaging. The mean gestational age in controls was 30.7 (SD, 4.2) weeks. At 28 weeks, fetuses with congenital diaphragmatic hernia had abnormal qualitative and quantitative maturation, more extra-axial fluid, and larger total skull volume. By 33 weeks, qualitative grading scores were still abnormal, but quantitative scoring was in the normal range. In contrast, the extra-axial fluid volume remained abnormal with increased ventricular volume. Normal brain parenchymal volumes were found. CONCLUSIONS: Brain development in fetuses with congenital diaphragmatic hernia around 28 weeks appears to be delayed. This feature is less prominent at 33 weeks. At this stage, there was also an increase in ventricular and extra-axial space volume.

AB - BACKGROUND AND PURPOSE: Congenital diaphragmatic hernia is associated with high mortality and morbidity, including evidence suggesting neurodevelopmental comorbidities after birth. The aim of this study was to document longitudinal changes in brain biometry and the cortical folding pattern in fetuses with congenital diaphragmatic hernia compared with healthy fetuses. MATERIALS AND METHODS: This is a retrospective cohort study including fetuses with isolated congenital diaphragmatic hernia between January 2007 and May 2019, with at least 2 MR imaging examinations. For controls, we used images from fetuses who underwent MR imaging for an unrelated condition that did not compromise fetal brain development and fetuses from healthy pregnant women. Biometric measurements and 3D segmentations of brain structures were used as well as qualitative and quantitative grading of the supratentorial brain. Brain development was correlated with disease-severity markers. RESULTS: Forty-two fetuses were included, with a mean gestational age at first MR imaging of 28.0 (SD, 2.1) weeks and 33.2 (SD, 1.3) weeks at the second imaging. The mean gestational age in controls was 30.7 (SD, 4.2) weeks. At 28 weeks, fetuses with congenital diaphragmatic hernia had abnormal qualitative and quantitative maturation, more extra-axial fluid, and larger total skull volume. By 33 weeks, qualitative grading scores were still abnormal, but quantitative scoring was in the normal range. In contrast, the extra-axial fluid volume remained abnormal with increased ventricular volume. Normal brain parenchymal volumes were found. CONCLUSIONS: Brain development in fetuses with congenital diaphragmatic hernia around 28 weeks appears to be delayed. This feature is less prominent at 33 weeks. At this stage, there was also an increase in ventricular and extra-axial space volume.

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DO - 10.3174/ajnr.A7760

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C2 - 36657946

AN - SCOPUS:85147318152

SN - 0195-6108

VL - 44

SP - 205

EP - 211

JO - AJNR. American journal of neuroradiology

JF - AJNR. American journal of neuroradiology

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ER -

Longitudinal MRI Evaluation of Brain Development in Fetuses with Congenital Diaphragmatic Hernia around the Time of Fetal Endotracheal Occlusion

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