TY - JOUR
T1 - Neurodevelopment in patients with biliary atresia up to toddler age
T2 - Outcomes and predictability
AU - Dibbits, Marloes H.J.
AU - Rodijk, Lyan H.
AU - den Heijer, Anne E.
AU - Bos, Arend F.
AU - Verkade, Henkjan J.
AU - de Kleine, Ruben H.
AU - Alizadeh, Behrooz Z.
AU - Hulscher, Jan B.F.
AU - Bruggink, Janneke L.M.
N1 - Funding Information:
Sponsored by a grant from the Dutch Digestive Foundation (Maag Lever Darm Stichting, MLDS), grant number JR/2016-098 . L.H. Rodijk was financially supported by a MD/PhD scholarship from the University Medical Center Groningen , Groningen, the Netherlands.
Publisher Copyright:
© 2023
PY - 2023/5
Y1 - 2023/5
N2 - Aim: To assess neurodevelopment in young patients with biliary atresia (BA) and to determine the predictive value of General Movement Assessment (GMA) at infant age for neurodevelopmental impairments at toddler age. Method: Infants diagnosed with BA were prospectively included in a longitudinal study. Neurodevelopmental status was previously assessed before Kasai porto-enterostomy (KPE) and one month after KPE using Prechtl's GMA, including motor optimality scores. At 2–3 years, neurodevelopment was assessed using the Bayley Scales of Infant Development, and compared to the Dutch norm population. The predictive value of GMA at infant age for motor skills and cognition at toddler age was determined. Results: Neurodevelopment was assessed in 41 BA patients. At toddler age (n = 38, age 29 ± 5 months, 70 % liver transplantation), 13 (39 %) patients scored below-average on motor skills, and 6 (17 %) patients on cognition. Abnormal GMA after KPE predicted both below-average motor skills and cognitive score at toddler age (sensitivity, 91 % and 80 %; specificity 83 % and 67 %; negative predictive value, 94 % and 94 %; and, positive predictive value, 77 % and 33 %, resp.). Interpretation: One-third of toddlers with BA show impaired motor skills. GMA post-KPE has a high predictive value to identify infants with BA at risk of neurodevelopmental impairments.
AB - Aim: To assess neurodevelopment in young patients with biliary atresia (BA) and to determine the predictive value of General Movement Assessment (GMA) at infant age for neurodevelopmental impairments at toddler age. Method: Infants diagnosed with BA were prospectively included in a longitudinal study. Neurodevelopmental status was previously assessed before Kasai porto-enterostomy (KPE) and one month after KPE using Prechtl's GMA, including motor optimality scores. At 2–3 years, neurodevelopment was assessed using the Bayley Scales of Infant Development, and compared to the Dutch norm population. The predictive value of GMA at infant age for motor skills and cognition at toddler age was determined. Results: Neurodevelopment was assessed in 41 BA patients. At toddler age (n = 38, age 29 ± 5 months, 70 % liver transplantation), 13 (39 %) patients scored below-average on motor skills, and 6 (17 %) patients on cognition. Abnormal GMA after KPE predicted both below-average motor skills and cognitive score at toddler age (sensitivity, 91 % and 80 %; specificity 83 % and 67 %; negative predictive value, 94 % and 94 %; and, positive predictive value, 77 % and 33 %, resp.). Interpretation: One-third of toddlers with BA show impaired motor skills. GMA post-KPE has a high predictive value to identify infants with BA at risk of neurodevelopmental impairments.
KW - Behavior
KW - Cognition
KW - IQ
KW - Liver disease
KW - Liver transplantation
KW - Motor skills
U2 - 10.1016/j.earlhumdev.2023.105754
DO - 10.1016/j.earlhumdev.2023.105754
M3 - Article
C2 - 37030125
AN - SCOPUS:85151564071
SN - 0378-3782
VL - 180
JO - Early Human Development
JF - Early Human Development
M1 - 105754
ER -