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Case: This rare case presents an isolated congenital shoulder dislocation in a twin delivery, without traumatic delivery. Delivered by emergent cesarean section at 33 weeks gestation, the infant presented with a lateral shoulder crease with x-rays showing anterior and inferior dislocation. Treatment included prompt reduction and stabilization, with follow-up ultrasound demonstrating a physeal injury.Conclusions: This case report presents the only published congenital shoulder dislocation in an infant after an atraumatic twin cesarean delivery. Prompt reduction, stabilization, and ultrasound imaging to assess for physeal injury is our recommended management for this scenario. Keywords: Neonatal shoulder dislocation, Congenital shoulder dislocation, Neonatal shoulder deformity