Osteofibrous dysplasia is a rare and benign disease that originates from the tibia or fibula. The symptoms of osteofibrous dysplasia include painless enlargement and bowing of the tibia and pain occurring in the presence of pathological fracture. Herein a male infant who was admitted with redness and swelling on the right leg and diagnosed as pathological tibia fracture due to left tibia osteofibrous dysplasia on the third day of life was presented. To our knowledge, this is the earliest presentation of osteofibrous dysplasia with a pathological fracture in a neonate. Therefore, it must be suspected in neonatal bone fractures.
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