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Early Motor Repertoire in Infants With Biliary Atresia: A Nationwide Prospective Cohort Study
Lyan H Rodijk1, Arend F Bos2, Henkjan J Verkade3
1Section of Pediatric Surgery, Department of Surgery.
Insights
Nearly half of infants with biliary atresia (BA) show atypical general movements (GMs) at diagnosis, indicating potential neurological impairment. Early assessment of GMs in BA infants is crucial for monitoring neurodevelopmental outcomes.
Area of Science:
- Neonatal neurology
- Developmental pediatrics
- Gastroenterology
Background:
- Biliary atresia (BA) is a serious neonatal liver disease.
- Neurological status in infants with BA is not well understood.
- General Movements (GMs) assessment is a validated tool for evaluating early neurological development.
Purpose of the Study:
- To assess the neurological status of infants diagnosed with biliary atresia (BA).
- To evaluate the utility of Prechtl's General Movement Assessment in infants with BA.
- To compare the motor repertoire of BA infants with healthy controls.
Main Methods:
- Prospective nationwide cohort study including infants with BA.
- Video recording and assessment of General Movements (GMs) and motor optimality scores (MOS) before Kasai portoenterostomy.
- Comparison of GM optimality scores and MOS with two reference groups of healthy infants.
Main Results:
- 46% of infants with BA (35 total) exhibited atypical GMs at diagnosis (PMA 47 weeks).
- Atypical GMs were significantly more prevalent in BA infants compared to healthy controls (46% vs. 10-18%, P < 0.001).
- No significant correlation was found between atypical GMs and clinical variables like bilirubin levels or anthropometric z-scores.
Conclusions:
- Atypical GMs are common in infants with BA at diagnosis, suggesting potential neurological impairment.
- Early detection of neurological abnormalities through GM assessment is vital in BA.
- Close neurodevelopmental monitoring is recommended for infants with BA.
Objectives:
The aim of the study was to assess the neurological status in infants with biliary atresia (BA) at time of diagnosis, using Prechtl's validated General Movement Assessment.
Methods:
Infants diagnosed with BA were prospectively included in a nationwide cohort study. From birth to approximately 46 weeks of postmenstrual age (PMA), general movements (GMs) are defined as "writhing movements." At 46 to 49 weeks PMA, "'fidgety movements" emerge. The infant's early motor repertoire was recorded on video before Kasai portoenterostomy. We scored GM optimality scores (min-max 5-42) or motor optimality scores (MOS, min-max 5-28) as appropriate. We defined GM optimality scores <36 and MOS <26 as atypical, and compared the results with 2 reference groups of healthy peers.
Results:
We assessed GMs in 35 infants with BA (11/35 boys, gestational age 40 weeks [36-42], birth weight 3370 g [2015-4285]). At time of diagnosis (PMA 47 weeks [42-60]), 16 infants (46%) showed atypical GMs. The proportion of infants with atypical GMs was significantly higher in BA (46%) than in 2 reference groups of healthy infants (vs 10%, P < 0.001; vs 18%, P < 0.001). Total and direct bilirubin levels were 165 μmol/L (87-364) and 134 μmol/L (72-334), respectively, height z score was 0.05 (-2.90, 1.75), weight z score -0.52 ([-2.50, -0.20) and mean upper arm circumference z score -1.80 (-2.50, -0.20). We found no statistically significant relation between atypical GMs and clinical variables.
Conclusions:
Almost half of the infants with BA showed atypical GMs at time of diagnosis, suggesting neurological impairment. Close monitoring of these infants is warranted to determine their individual neurodevelopmental trajectories.
