Background:It is a rare condition defined by congenital pseudarthrosis of the fibula (CPF), which may be associated with neurofibromatosis (NF) and congenital pseudarthrosis of the tibia. A condition such as this may be symptomatic or asymptomatic, but it requires treatment because of the progression of signs such as ankle valgus and pseudarthrosis of the tibia. There are a number of surgical techniques that can be used to treat this condition, such as tibiofibular fusion, internal fixation, the Ilizarov method, and periosteal flap.Aim:The goal of this study was to describe the treatment results of two patients with CPF using vascularized fibular periosteal flaps.Case report:We described the case of a 5-year-old patient and a 19-month-old patient with isolated CPF. Both patients underwent a distal-based vascularized fibular periosteal flap, and intramedullary fixation was used to treat the patients.Conclusion:The patients had full union in the pseudarthrosis site, but in the end, both had asymptomatic refracture in the union site. Our experiences showed that it is necessary to use strong intramedullary fixation and bone graft.