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PEDIATRICS Vol. 113 No. 6 June 2004, pp. 1549-1558

Cognitive Function of Children With Cystic Fibrosis: Deleterious Effect of Early Malnutrition

Rebecca L. Koscik, PhD*, Philip M. Farrell, MD, PhD{ddagger}, Michael R. Kosorok, PhD*, Kathleen M. Zaremba, MPH{ddagger}, Anita Laxova, BS{ddagger}, Hui-Chuan Lai, PhD§, Jeff A. Douglas, PhD||, Michael J. Rock, MD{ddagger} and Mark L. Splaingard, MD

* Department of Biostatistics/Medical Informatics, University of Wisconsin; Madison, Wisconsin
{ddagger} Department of Pediatrics, University of Wisconsin; Madison, Wisconsin
§ Department of Nutritional Sciences, University of Wisconsin; Madison, Wisconsin
|| Department of Statistics, University of Illinois at Urbana-Champaign, Urbana-Champaign, Illinois
Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin

Objective. Patients who have cystic fibrosis (CF) and experience delayed diagnosis by traditional methods have greater nutritional insult compared with peers diagnosed via neonatal screening. The objective of this study was to evaluate cognitive function in children with CF and the influence of both early diagnosis through neonatal screening and the potential effect of early malnutrition.

Methods. Cognitive assessment data were obtained for 89 CF patients (aged 7.3-17 years) during routine clinic visits. Patients had been enrolled in either the screened (N = 42) or traditional diagnosis (control) group (N = 47) of the Wisconsin CF Neonatal Screening Project. The Test of Cognitive Skills, Second Edition was administered to generate the Cognitive Skills Index (CSI) and cognitive factor scores (Verbal, Nonverbal, and Memory).

Results. Cognitive scores in the overall study population were similar to normative data (CSI mean [standard deviation]: 102.5 [16.6]; 95% confidence interval: 99.1-105.9). The mean (standard deviation) CSI scores for the screened and control groups were 104.4 (14.4) and 99.8 (18.5), respectively. Significantly lower cognitive scores correlated with indicators of malnutrition and unfavorable family factors such as single parents, lower socioeconomic status, and less parental education. Our analyses revealed lower cognitive scores in patients with low plasma {alpha}-tocopherol ({alpha}-T) levels at diagnosis. In addition, patients in the control group who also had vitamin E deficiency at diagnosis ({alpha}-T < 300 µg/dl) showed significantly lower CSI scores in comparison with {alpha}-T–sufficient control subjects and both deficient and sufficient {alpha}-T subsets of screened patients.

Conclusion. Results suggest that prevention of prolonged malnutrition by early diagnosis and nutritional therapy, particularly minimizing the duration of vitamin E deficiency, is associated with better cognitive functioning in children with CF.

Key Words: cognitive function • cystic fibrosis • malnutrition • neonatal screening • vitamin E deficiency

Abbreviations: CF, cystic fibrosis • SES, socioeconomic status • {alpha}-T, {alpha}-tocopherol • TCS/2, Test of Cognitive Skills, Second Edition • CSI, Cognitive Skills Index • MI, meconium ileus • PS, pancreatic sufficient • PI, pancreatic insufficient

Received for publication Jun 2, 2003; Accepted Oct 20, 2003.




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