Early motor outcomes in infants with critical congenital heart disease are related to neonatal brain development and brain injury

• Published in: Developmental medicine and child neurology Vol. 64; no. 2; pp. 192 - 199
• Main Authors: Stegeman, Raymond, Sprong, Maaike C A, Breur, Johannes M P J, Groenendaal, Floris, Vries, Linda S, Haas, Felix, Net, Janjaap, Jansen, Nicolaas J G, Benders, Manon J N L, Claessens, Nathalie H P, Toet, Mona C, Dudink, Jeroen, Gronden, Charlotte M A, Flamman, Wiebe R, Zwart, Sophie A, Slieker, Martijn G, Nijman, Joppe, Wösten‐van Asperen, Roelie M, Schappin, Renske, Uniken Venema‐van Uden, Monica M A, Heus, Roel
• Format: Journal Article
• Language: English
• Published: England John Wiley and Sons Inc 01-02-2022
• Subjects: Brain Injuries - diagnostic imaging; Brain Injuries - pathology; Brain Injuries - physiopathology; Cerebellum - diagnostic imaging; Cerebellum - growth & development; Cerebellum - pathology; Cerebral Cortex - diagnostic imaging; Cerebral Cortex - growth & development; Cerebral Cortex - pathology; Cerebral Palsy - diagnostic imaging; Cerebral Palsy - pathology; Cerebral Palsy - physiopathology; Child Development - physiology; Developmental Disabilities - diagnostic imaging; Developmental Disabilities - etiology; Developmental Disabilities - pathology; Developmental Disabilities - physiopathology; Female; Gray Matter - diagnostic imaging; Gray Matter - growth & development; Gray Matter - pathology; Heart Defects, Congenital - complications; Heart Defects, Congenital - diagnostic imaging; Heart Defects, Congenital - surgery; Humans; Infant; Infant, Newborn; Ischemic Stroke - diagnostic imaging; Ischemic Stroke - pathology; Ischemic Stroke - physiopathology; Magnetic Resonance Imaging; Male; Motor Skills - physiology; Original; Prospective Studies; Pyramidal Tracts - diagnostic imaging; Pyramidal Tracts - growth & development; Pyramidal Tracts - pathology; White Matter - diagnostic imaging; White Matter - growth & development; White Matter - pathology
• ISSN: 0012-1622; 1469-8749
• DOI: 10.1111/dmcn.15024

Aim
To assess the relationship between neonatal brain development and injury with early motor outcomes in infants with critical congenital heart disease (CCHD).
Method
Neonatal brain magnetic resonance imaging was performed after open‐heart surgery with cardiopulmonary bypass. Cortical grey matter (CGM), unmyelinated white matter, and cerebellar volumes, as well as white matter motor tract fractional anisotropy and mean diffusivity were assessed. White matter injury (WMI) and arterial ischaemic stroke (AIS) with corticospinal tract (CST) involvement were scored. Associations with motor outcomes at 3, 9, and 18 months were corrected for repeated cardiac surgery.
Results
Fifty‐one infants (31 males, 20 females) were included prospectively. Median age at neonatal surgery and postoperative brain magnetic resonance imaging was 7 days (interquartile range [IQR] 5–11d) and 15 days (IQR 12–21d) respectively. Smaller CGM and cerebellar volumes were associated with lower fine motor scores at 9 months (CGM regression coefficient=0.51, 95% confidence interval [CI]=0.15–0.86; cerebellum regression coefficient=3.08, 95% CI=1.07–5.09) and 18 months (cerebellum regression coefficient=2.08, 95% CI=0.47–5.12). The fractional anisotropy and mean diffusivity of white matter motor tracts were not related with motor scores. WMI was related to lower gross motor scores at 9 months (mean difference −0.8SD, 95% CI=−1.5 to −0.2). AIS with CST involvement increased the risk of gross motor problems and muscle tone abnormalities. Cerebral palsy (n=3) was preceded by severe ischaemic brain injury.
Interpretation
Neonatal brain development and injury are associated with fewer favourable early motor outcomes in infants with CCHD.