Neurodevelopmental outcome of infants with congenital diaphragmatic hernia prospectively enrolled in an interdisciplinary follow-up program

被引:77
作者
Danzer, Enrico
Gerdes, Marsha
Bernbaum, Judy
D'Agostino, Joann
Bebbington, Michael W.
Siegle, Jennifer
Hoffman, Casey
Rintoul, Natalie E.
Flake, Alan W.
Adzick, N. Scott
Hedrick, Holly L. [1 ]
机构
[1] Childrens Hosp Philadelphia, Ctr Fetal Diag & Treatment, Philadelphia, PA 19104 USA
关键词
Congenital diaphragmatic hernia; Neurodevelopmental outcome; Bayley Scales of Infant Development; Wechsler Preschool and Primary Scale of Intelligence; Liver position; Extracorporeal membrane oxygenation; EXTRACORPOREAL MEMBRANE-OXYGENATION; PRESCHOOL DIAGNOSIS; EARLY MOTOR; SCHOOL-AGE; CHILDREN; SURVIVORS; LANGUAGE; RISK; MANAGEMENT; REPAIR;
D O I
10.1016/j.jpedsurg.2010.03.011
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Purpose: The purpose of the study was to evaluate the neurodevelopmental outcome in infants with congenital diaphragmatic hernia (CDH). Methods: Between June 2004 and September 2007, 41 CDH survivors were prospectively enrolled in an interdisciplinary follow-up program. Neurodevelopmental status was evaluated using the Bayley Scales of Infant Development II (prior 2006, n = 9), the Bayley Scales of Infant Development III (after 2006, n = 27), or the Wechsler Preschool and Primary Scale of Intelligence III (children older than 4 years, n = 5). Scores were grouped as average, mildly delayed, and severely delayed by standard deviation intervals (115-85, 71-84, <70), and mixed if average and mildly delayed in either cognitive or language. Results: Median age at last assessment was 24 months (range, 6-62). Average, mixed, mildly delayed, and severely delayed scores for neurocognitive and language skills were found in 49%, 19%, 17%, and 15%, respectively. Psychomotor scores were normal, mildly delayed, and severely delayed in 46%, 23%, and 31%, respectively. Autism was present in 7%. Abnormal muscle tonicity was found in 51% (49% hypotonic, 2% hypertonic). Multivariate risk factors for borderline or delayed neurodevelopmental, neurocognitive, and/or psychomotor outcome were intrathoracic liver position (P =.02), presence of a right-sided CDH (P =.02), extracorporeal membrane oxygenation need (P <.001), Gore-Tex patch repair (P =.02), O-2 requirement at 30 days of life (P <.01), and hypotonicity (P <.01). Conclusions: The prospective evaluation in an interdisciplinary follow-up program uncovered striking morbidities in neurodevelopmental status in approximately half of the CDH infants. The most common neurologic sequelae are neuromuscular hypotonicity and psychomotor dysfunction. Patient-specific factors are important determinants of adverse neurologic outcome. (C) 2010 Published by Elsevier Inc.
引用
收藏
页码:1759 / 1766
页数:8
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