Combination Treatment by Cross-Union of the Tibia and Fibula, Autogenic Iliac Bone Grafting, Reliable Fixation and Bone Morphogenetic Proteins for the Treatment of Refractory Congenital Pseudarthrosis of the Tibia

Abstract

Background: The treatment of congenital pseudarthrosis of the tibia (CPT) remains a challenge because of the difficulties of achieving and maintaining bone union, as well as complications of joint deformity and limb-length discrepancy. The purpose of this study was to evaluate the efficacy of cross-union of the tibia and fibula in achieving union and preventing refracture for patients with refractory CPT as a complementary approach to improve upon conventional surgical treatments.

Methods: A retrospective study including patients with refractory CPT who attended our department between June 2014 and August 2020. Eighteen CPT patients, who had sustained refracture that required cast immobilization or secondary surgery, and were managed by pseudarthrosis resection, cross-union of the tibia and fibula, bone morphogenetic protein-2 and autogenous iliac bone grafting, were included. Clinical outcomes of the bone union rate and the frequency of refracture after performing cross-union of the tibia and fibula were assessed during the follow-up period.

Results: The mean follow-up period was 4.3 years (range: 1.5 to 6.25 y). The mean age of the patients at surgery was 5.4 years (range: 2.6 to 10 y), and all 18 (100%) of the 18 patients had final healing at the site of pseudarthrosis. The average time spent to achieve radiologic bone union of the pseudarthrosis after operation was 2.96 months (range: 2.2 to 4.1 mo). Two (11.1%) patients had an average 2.5 cm limb-length discrepancy, none (0%) sustained refracture which needed cast immobilization or secondary surgery. Patients were all pain-free and move actively.

Conclusions: Cross-union of the tibia and fibula is a promising complementary procedure for treating refractory CPT patients.

Level of evidence: Level IV-case series.

FIGURE 1

A, Exposure of the tibia, interosseous membrane and fibula; resection of the membrane under direct vision; circumferential resection of the tibial fibrous hamartoma at the planned length of the cross-union. B, A periosteal graft was harvested from the undersurface of the iliacus muscle. It was then expanded by passing it through the skin graft mesher. C, An appropriate length and diameter intramedullary rod and locking compression plate system were used to immobilize the tibia; the periosteal graft was wrapped around the congenital pseudarthrosis site and bone morphogenic protein-2 collagen sponges were inserted over the posterior muscles behind the tibia and fibula (left). The cancellous bone was inserted between the tibia and fibula (left center). Bone morphogenic protein-2 sponges were placed over the bone graft (right center).

FIGURE 2

Case 1. A child aged 5 years and 1 month with congenital pseudarthrosis of the tibia, had been treated by traditional surgery with pseudarthrosis resection, autologous iliac bone allografting, and intramedullary nail fixation before treating by cross-union of the tibia and fibula. The first procedure was a failure with bone nonunion and lower limb deformity. Three years later, we treated the child by cross-union of the tibia and fibula. Nearly 2.3 months after the second surgery, we found that the child had obtained initial bone union of the pseudarthrosis. One year after the second surgery, we found the child recovered well without lower limb deformity. Anteroposterior (right) and lateral (left) radiographs of (A) preoperation, (B) 4 days after the first surgery, (C) 3 years after the first surgery, (D) treating by cross-union of the tibia and fibula, (E) 2.3 months after the second surgery, (F) 1 year after the second surgery.

FIGURE 3

Case 2. A child aged 5 years and 2 months with congenital pseudarthrosis of the tibia, had undergone traditional surgery twice with pseudarthrosis resection, autologous iliac bone allografting and intramedullary nail fixation before treating by cross-union of the tibia and fibula. The first 2 surgeries were failures with bone nonunion and lower limb deformities. We treated the child with the cross-union of the tibia and fibula procedure. Nearly 3.2 months after the third surgery, we found that the child had obtained initial bone union of the pseudarthrosis. Seven months after the third surgery, we found the child lower limb deformity with 2 cm limb-length discrepancy. Anteroposterior (right) and lateral (left) radiographs of (A) preoperation, (B) the first surgery, (C) 4 months after the first surgery, (D) the second surgery, (E) 2 years after the second surgery, (F) the third surgery, (G) 3.2 months after the third surgery, (H) 7 months after the third surgery.