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Congenital radial head dislocation is a rare anomaly of the pediatric musculoskeletal system. The most common type is posterior, with anterior and lateral dislocations being less common. Unilateral cases are even more uncommon and were earlier thought to be non-existent. Our case report describes a young girl with unilateral congenital anterior radial head dislocation with progressive development of cubitus valgus deformity, which is a very rare occurrence. On reviewing literature, only few such cases have been described. Our patient also had a history of developmental dysplasia of hip and a congenital intra-hepatic porto-systemic shunt, which in addition to the morphological appearances of the radial head and capitellum, suggested a congenital etiology.