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Abstract Purpose  The purpose was to report a case of elevated intraocular vascular endothelial growth factor (VEGF) levels in a 2-year-old boy with stage 4 Coats disease. Design  Interventional case report. Methods  A 2-year-old boy with severe Coats disease and complex retinal detachment refractory to surgical intervention underwent two injections of intravitreal pegaptanib sodium in his right eye 8 weeks apart. Results  After intravitreal injection there was a marked reduction in VEGF (908 to 167 pg/ml) levels and improvement of exudation, hemorrhage, and nearly complete reattachment of his retina within 3 weeks of treatment. The patient remains stable 6 months post-treatment. Conclusions  Coats disease may have a component of dysregulation of VEGF-mediated angiogenesis as evidenced by elevated levels in this patient. [ABSTRACT FROM AUTHOR]