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Preschool neurological assessment in congenital diaphragmatic hernia survivors: outcome and perinatal factors associated with neurodevelopmental impairment.

Abstract：

OBJECTIVE:To evaluate the preschool neurodevelopmental outcome in CDH survivors between 2 and 5 years of age and to identify perinatal and postnatal factors that may be predictive of persistent neurological deficits. METHODS:The study cohort consists of 60 CDH survivors that were enrolled in our prospective, follow-up program between January 2006 and September 2012. Developmental assessment of study participants between 2 and 3 years of age was performed using the Bayley Scales of Infant Development, 3rd Edition (BSID-III). Cognitive outcomes of CDH children older than 3 years of age were evaluated using The Wechsler Preschool and Primary Scale of Intelligence, 3rd Edition (WPPSI-III). Neurodevelopmental delay was defined by a score of ≤ 85 in any of the evaluated composite scores. Severe impairments were defined as a score of ≤ 69 in at least one domain tested. RESULTS:Mean follow-up for children assessed by BSID-III (n=42) and WPPSI-III (n=18) was 28 ± 4.5 months and 58 ± 4.0 months, respectively. As a group, mean composite and subdomain BSID-III and WPPSI-III scores were within the expected normal range. For the BSID-III group, 9 (22%) had mild deficits in at least one domain, and 6 (14%) had severe deficits in at least one. Only 3 (7%) patients demonstrated severe neurological delays for all BSID-III composite scales. For the WPPSI-III group, 4 (22%) patients scored within the borderline-delayed range for at least one subscale and only one (6%) child had a WPPSI-III VIQ score in the severe delay range. Abnormal muscle tonicity was found in 35% (hypotonicity 33%, hypertonicity 2%). Need for ECMO, prolonged ventilation, hypotonicity, and other surrogate markers of disease severity (P<0.05) were associated with borderline or delayed neurological outcome. CONCLUSION:The majority of CDH children are functioning in the average range at early preschool and preschool age. Neuromuscular hypotonicity is common in CDH survivors. CDH severity appears to be predictive of adverse neurodevelopmental outcome.

journal_name

Early Hum Dev

journal_title

Early human development

authors

Danzer E,Gerdes M,D'Agostino JA,Partridge EA,Hoffman-Craven CH,Bernbaum J,Rintoul NE,Flake AW,Adzick NS,Hedrick HL

doi

10.1016/j.earlhumdev.2012.12.009

subject

Has Abstract

pub_date

2013-06-01 00:00:00

pages

393-400

issue

eissn

0378-3782

issn

1872-6232

pii

S0378-3782(12)00293-9

journal_volume

pub_type

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Preschool neurological assessment in congenital diaphragmatic hernia survivors: outcome and perinatal factors associated with neurodevelopmental impairment.