Association of genetic and sulcal traits with executive function in congenital heart disease.

Autor: Maleyeff L; Department of Biostatistics, Harvard T.H. Chan School of Public Health, Boston, Massachusetts, USA., Newburger JW; Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.; Department of Cardiology, Boston Children's Hospital, Boston, Massachusetts, USA., Wypij D; Department of Biostatistics, Harvard T.H. Chan School of Public Health, Boston, Massachusetts, USA.; Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.; Department of Cardiology, Boston Children's Hospital, Boston, Massachusetts, USA., Thomas NH; Department of Child and Adolescent Psychiatry and Behavioral Sciences and Center for Human Phenomic Science, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.; Department of Psychiatry, University of Pennsylvania, Philadelphia, Pennsylvania, USA., Anagnoustou E; Department of Pediatrics, Holland Bloorview Kids Rehabilitation Hospital, University of Toronto, Toronto, Ontario, Canada., Brueckner M; Department of Genetics, Yale University School of Medicine, New Haven, Connecticut, USA.; Department of Pediatrics, Yale University School of Medicine, New Haven, Connecticut, USA., Chung WK; Department of Pediatrics, Columbia University Medical Center, New York, New York, USA.; Department of Medicine, Columbia University Medical Center, New York, New York, USA., Cleveland J; Department of Surgery, Keck School of Medicine, University of Southern California, Los Angeles, California, USA.; Department of Pediatrics, Keck School of Medicine, University of Southern California, Los Angeles, California, USA., Cunningham S; Division of General Pediatrics, Department of Pediatrics, University of Utah, Salt Lake City, Utah, USA., Gelb BD; Mindich Child Health and Development Institute and Department of Pediatrics, Icahn School of Medicine at Mount Sinai, New York, New York, USA., Goldmuntz E; Division of Cardiology, Department of Pediatrics, Children's Hospital of Philadelphia, Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania, USA., Hagler DJ Jr; Center for Multimodal Imaging and Genetics, University of California San Diego, San Diego, California, USA.; Department of Radiology, School of Medicine, University of California San Diego, San Diego, California, USA., Huang H; Department of Radiology, Children's Hospital of Philadelphia, University of Pennsylvania, Philadelphia, Pennsylvania, USA., King E; Department of Pediatrics, University of Cincinnati, Cincinnati, Ohio, USA.; Division of Biostatistics and Epidemiology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA., McQuillen P; Department of Pediatrics, University of California, San Francisco, California, USA.; Department of Neurology, University of California, San Francisco, California, USA., Miller TA; Department of Pediatrics, Primary Children's Hospital, University of Utah, Salt Lake City, Utah, USA.; Division of Pediatric Cardiology, Maine Medical Center, Portland, Maine, USA., Norris-Brilliant A; Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA., Porter GA Jr; Department of Pediatrics, University of Rochester Medical Center, Rochester, New York, USA., Roberts AE; Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.; Department of Cardiology, Boston Children's Hospital, Boston, Massachusetts, USA.; Division of Genetics and Genomics, Boston Children's Hospital, Boston, Massachusetts, USA., Grant PE; Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.; Division of Newborn Medicine, Department of Pediatrics, Boston Children's Hospital, Boston, Massachusetts, USA.; Fetal Neonatal Neuroimaging and Developmental Science Center, Boston Children's Hospital, Boston, Massachusetts, USA.; Department of Radiology, Boston Children's Hospital, Boston, Massachusetts, USA., Im K; Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.; Division of Newborn Medicine, Department of Pediatrics, Boston Children's Hospital, Boston, Massachusetts, USA.; Fetal Neonatal Neuroimaging and Developmental Science Center, Boston Children's Hospital, Boston, Massachusetts, USA., Morton SU; Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.; Division of Newborn Medicine, Department of Pediatrics, Boston Children's Hospital, Boston, Massachusetts, USA.; Fetal Neonatal Neuroimaging and Developmental Science Center, Boston Children's Hospital, Boston, Massachusetts, USA.
Jazyk: angličtina
Zdroj: Annals of clinical and translational neurology [Ann Clin Transl Neurol] 2024 Feb; Vol. 11 (2), pp. 278-290. Date of Electronic Publication: 2023 Nov 27.
DOI: 10.1002/acn3.51950
Abstrakt: Objective: Persons with congenital heart disease (CHD) are at increased risk of neurodevelopmental disabilities, including impairments to executive function. Sulcal pattern features correlate with executive function in adolescents with single-ventricle heart disease and tetralogy of Fallot. However, the interaction of sulcal pattern features with genetic and participant factors in predicting executive dysfunction is unknown.
Methods: We studied sulcal pattern features, participant factors, and genetic risk for executive function impairment in a cohort with multiple CHD types using stepwise linear regression and machine learning.
Results: Genetic factors, including predicted damaging de novo or rare inherited variants in neurodevelopmental disabilities risk genes, apolipoprotein E genotype, and principal components of sulcal pattern features were associated with executive function measures after adjusting for age at testing, sex, mother's education, and biventricular versus single-ventricle CHD in a linear regression model. Using regression trees and bootstrap validation, younger participant age and larger alterations in sulcal pattern features were consistently identified as important predictors of decreased cognitive flexibility with left hemisphere graph topology often selected as the most important predictor. Inclusion of both sulcal pattern and genetic factors improved model fit compared to either alone.
Interpretation: We conclude that sulcal measures remain important predictors of cognitive flexibility, and the model predicting executive outcomes is improved by inclusion of potential genetic sources of neurodevelopmental risk. If confirmed, measures of sulcal patterning may serve as early imaging biomarkers to identify those at heightened risk for future neurodevelopmental disabilities.
(© 2023 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.)
Databáze: MEDLINE
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