TY - JOUR
T1 - Predictive and diagnostic measures for kernicterus spectrum disorder
T2 - a prospective cohort study
AU - Gelineau-Morel, Rose
AU - Usman, Fatima
AU - Shehu, Saadatu
AU - Yeh, Hung Wen
AU - Suwaid, Mohammad A.
AU - Abdulsalam, Mohammed
AU - Jibril, Yasir
AU - Satrom, Katherine M.
AU - Shapiro, Steven M.
AU - Zinkus, Timothy P.
AU - Head, Hayden W.
AU - Slusher, Tina M.
AU - Le Pichon, Jean Baptiste
AU - Farouk, Zubaida L.
N1 - Publisher Copyright:
© 2023, The Author(s), under exclusive licence to the International Pediatric Research Foundation, Inc.
PY - 2024/1
Y1 - 2024/1
N2 - Background: Kernicterus spectrum disorder (KSD) resulting from neonatal hyperbilirubinemia remains a common cause of cerebral palsy worldwide. This 12-month prospective cohort study followed neonates with hyperbilirubinemia to determine which clinical measures best predict KSD. Methods: The study enrolled neonates ≥35 weeks gestation with total serum bilirubin (TSB) ≥ 20 mg/dl admitted to Aminu Kano Hospital, Nigeria. Clinical measures included brain MRI, TSB, modified bilirubin-induced neurologic dysfunction (BIND-M), Barry-Albright Dystonia scale (BAD), auditory brainstem response (ABR), and the modified KSD toolkit. MRI signal alteration of the globus pallidus was scored using the Hyperbilirubinemia Imaging Rating Tool (HIRT). Results: Of 25 neonates enrolled, 13/25 completed 12-month follow-up and six developed KSD. Neonatal BIND-M ≥ 3 was 100% sensitive and 83% specific for KSD. Neonatal ABR was 83% specific and sensitive for KSD. Neonatal HIRT score of 2 was 67% sensitive and 75% specific for KSD; this increased to 100% specificity and sensitivity at 12 months. BAD ≥ 2 was 100% specific for KSD at 3–12 months, with 50–100% sensitivity. Conclusions: Neonatal MRIs do not reliably predict KSD. BIND-M is an excellent screening tool for KSD, while the BAD or HIRT score at 3 or 12 months can confirm KSD, allowing for early diagnosis and intervention. Impact: The first prospective study of children with acute bilirubin encephalopathy evaluating brain MRI findings over the first year of life.Neonatal MRI is not a reliable predictor of kernicterus spectrum disorders (KSD).Brain MRI at 3 or 12 months can confirm KSD.The modified BIND scale obtained at admission for neonatal hyperbilirubinemia is a valuable screening tool to assess risk for developing KSD.The Barry Albright Dystonia scale and brain MRI can be used to establish a diagnosis of KSD in at-risk infants as early as 3 months.
AB - Background: Kernicterus spectrum disorder (KSD) resulting from neonatal hyperbilirubinemia remains a common cause of cerebral palsy worldwide. This 12-month prospective cohort study followed neonates with hyperbilirubinemia to determine which clinical measures best predict KSD. Methods: The study enrolled neonates ≥35 weeks gestation with total serum bilirubin (TSB) ≥ 20 mg/dl admitted to Aminu Kano Hospital, Nigeria. Clinical measures included brain MRI, TSB, modified bilirubin-induced neurologic dysfunction (BIND-M), Barry-Albright Dystonia scale (BAD), auditory brainstem response (ABR), and the modified KSD toolkit. MRI signal alteration of the globus pallidus was scored using the Hyperbilirubinemia Imaging Rating Tool (HIRT). Results: Of 25 neonates enrolled, 13/25 completed 12-month follow-up and six developed KSD. Neonatal BIND-M ≥ 3 was 100% sensitive and 83% specific for KSD. Neonatal ABR was 83% specific and sensitive for KSD. Neonatal HIRT score of 2 was 67% sensitive and 75% specific for KSD; this increased to 100% specificity and sensitivity at 12 months. BAD ≥ 2 was 100% specific for KSD at 3–12 months, with 50–100% sensitivity. Conclusions: Neonatal MRIs do not reliably predict KSD. BIND-M is an excellent screening tool for KSD, while the BAD or HIRT score at 3 or 12 months can confirm KSD, allowing for early diagnosis and intervention. Impact: The first prospective study of children with acute bilirubin encephalopathy evaluating brain MRI findings over the first year of life.Neonatal MRI is not a reliable predictor of kernicterus spectrum disorders (KSD).Brain MRI at 3 or 12 months can confirm KSD.The modified BIND scale obtained at admission for neonatal hyperbilirubinemia is a valuable screening tool to assess risk for developing KSD.The Barry Albright Dystonia scale and brain MRI can be used to establish a diagnosis of KSD in at-risk infants as early as 3 months.
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U2 - 10.1038/s41390-023-02810-z
DO - 10.1038/s41390-023-02810-z
M3 - Article
C2 - 37689774
AN - SCOPUS:85170059300
SN - 0031-3998
VL - 95
SP - 285
EP - 292
JO - Pediatric Research
JF - Pediatric Research
IS - 1
ER -