4.7 Article

Visuomotor Performance in KCNJ11-Related Neonatal Diabetes Is Impaired in Children With DEND-Associated Mutations and May Be Improved by Early Treatment With Sulfonylureas

Journal

DIABETES CARE
Volume 35, Issue 10, Pages 2086-2088

Publisher

AMER DIABETES ASSOC
DOI: 10.2337/dc11-2225

Keywords

-

Funding

  1. Juvenile Diabetes Research Foundation [9-2008-177]
  2. American Diabetes Association [1-11-CT-41]
  3. National Institutes of Health (NIH) [P60-DK-020595, UL1-RR-024999]
  4. Comer Scholar
  5. Health Resources and Services Administration of the U.S. Department of Health and Human Services [T 73 MC11047]
  6. NIH/the Eunice Kennedy Shriver National Institute of Child Health and Human Development [P30-HD-054275]

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OBJECTIVE-To assess performance on an age-standardized neuromotor coordination task among sulfonylurea-treated KCNJ11-related neonatal diabetic patients. RESEARCH DESIGN AND METHODS-Nineteen children carrying KCNJ11 mutations associated with isolated diabetes (R201H; n = 8), diabetes with neurodevelopmental impairment (V59M or V59A [V59M/A]; n = 8), or diabetes not consistently associated with neurodevelopmental disability (Y330C, E322K, or R201C; n = 3) were studied using the age-standardized Beery-Buktenica Developmental Test of Visual-Motor Integration (VMI). RESULTS-Although R201H subjects tested in the normal range (median standard score = 107), children with V59M/A mutations had significantly lower than expected VMI standard scores (median = 49). The scores for all three groups were significantly different from each other (P = 0.0017). The age of sulfonylurea initiation was inversely correlated with VMI scores in the V59M/A group (P < 0.05). CONCLUSIONS-Neurodevelopmental disability in KCNJ11-related diabetes includes visuomotor problems that may be ameliorated by early sulfonylurea treatment. Comprehensive longitudinal assessment on larger samples will be imperative. Diabetes Care 35:2086-2088, 2012

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