Congenital pseudarthrosis of the tibia (CPT) is a rare pathology with reported incidence between 1:140,000 and 1:250,000. The natural history of the disease is extremely unfavourable and once a fracture occurs, there is a little or no tendency for the lesion to heal spontaneously. Histologically tibia shows area of segmental dysplasia resulting in anterolateral bowing of the bone Treatment is mainly surgical. The key to get primary union is to excise hamartomatous tissue and pathological

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... eum. Each child with CPT must be followed up till skeletal maturity to identify and rectify residual problems after primary healing. We are illustrating a case of severe neonatal form with posterior bowing at birth.