Abstract

BackgroundThe purpose of this study was to investigate the initial union rate, refracture rate and residual deformities of congenital pseudarthrosis of the tibia (CPT), using combined surgery including pseudarthrosis resection, intramedullary rodding, autogenous iliac bone grafting and Ilizarov’s fixator, with a mean 5.2 years follow-up.MethodsWe retrospectively reviewed the records and diagrams of patients with Crawford type IV congenital pseudarthrosis of the tibia between February 2007 and March 2010. Patients managed by pseudarthrosis resection, intramedullary rod of the tibia, wrapping autogenous iliac bone grafting and Ilizarov’s fixator were enrolled. We evaluated the bone union rate, tibial alignment, limb length discrepancy (LLD), valgus deformity of the ankle and the frequencies of refracture during period of follow-up.ResultsThere were 56 cases enrolled in the study, with a mean follow-up 5.2 years (range, 3 to 6.7 years). The mean age of the patients at surgery was 3.5 years (range, 1.5 to 12.4 years). Fifty (89.2 %) of the 56 patients had primary bone union at site of pseudarthrosis, while 5 obtained union after second surgery and 1 failed. The average time spent to obtain pseudarthrosis initial union was 4.5 months (range, 3.0 to 10.0 months) and mean duration of Ilizarov treatment was 4.7 months (range, 3.2 to 10.4 months). Eleven (19.6 %) patients had proximal tibial valgus with a mean angle of 9.5° (range, 5 to 24°), while 10 (17.9 %) patients had ankle valgus deformities with a mean of 12.3° (range, 6 to 21°). Sixteen (28.6 %) patients had an average 2.2 cm LLD (range, 1.5–4.2 cm). Of the 50 cases who obtained initial bone union of pseudarthrosis, 13 (26.0 %) had refracture which need cast immobilization or secondary surgery.ConclusionsThis combined surgery obtained initial union rate of 89.2 % at primary surgery while the refracture rate is 26.0 %. However, residual deformities such as proximal tibial valgus, LLD and ankle valgus were also existed which should be pay more attention to and dealt with.Trial registrationThis study was registered in ClinicalTrials.gov under the name “The Effect of Combined Surgery in Management of Congenital Pseudarthrosis of Tibia” (NCT02640040), which was released on August 31, 2015.

Highlights

  • The purpose of this study was to investigate the initial union rate, refracture rate and residual deformities of congenital pseudarthrosis of the tibia (CPT), using combined surgery including pseudarthrosis resection, intramedullary rodding, autogenous iliac bone grafting and Ilizarov’s fixator, with a mean 5.2 years follow-up

  • Congenital pseudarthrosis of the tibia (CPT) is a rare disorder and characterized by segmental osseous dysplasia and progression to fracture at walking age with a substantial risk of non-union, leg length discrepancy (LLD), and malalignment of the tibia

  • Intramedullary rod has been widely used for treatment of CPT to obtain union and protect against refracture, but it is an unstable fixation of the distal tibial fragment, and may cause ankle stiffness and arthritic changes in the ankle joint [8,9,10]

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Summary

Introduction

The purpose of this study was to investigate the initial union rate, refracture rate and residual deformities of congenital pseudarthrosis of the tibia (CPT), using combined surgery including pseudarthrosis resection, intramedullary rodding, autogenous iliac bone grafting and Ilizarov’s fixator, with a mean 5.2 years follow-up. CPT is recalcitrant to standard treatment and patients with this disorder often require multiple surgical procedures in an effort to achieve bone union and a functional extremity [1,2,3]. Three surgical techniques have been most commonly used, including vascularized fibular graft, intramedullary rod stabilization associated autogenous bone-grafting or bone morphogenetic protein-2, and Ilizarov technique, obtaining markedly improved primary union rate of CPT (range, 28 to 92 %) [4,5,6,7]. There is a risk of refracture after removal of the fixator [10,11,12]

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