Congenital Anterolateral Bowing Research Articles

Bilateral and symmetrical anteromedial bowing of the lower limbs in a patient with neurofibromatosis type-I.

An 8-year-old girl was referred to our department because of generalized bowing of long bones (radii, ulnae, and femora) and significant bilateral and symmetrical posteromedial bowing of the tibiae and fibulae. The femora were laterally bowed whereas the tibiae and fibulae showed posteromedial bowing between the middle and distal thirds of the tibia with posterior cortical thickening effectively causing the development of bilateral congenital anterolateral bowing of the tibiae and fibulae. We referred to closing-wedge osteotomy of the left tibia along with fibular osteotomy in order to realign the deformity. Due to the delayed appearance of skin stigmata in her early life, the diagnosis of neurofibromatosis was ruled out. At the age of 9 years, café-au-lait spots and axillary freckling were apparent. Genetic tests confirmed von Recklinghausen disease (neurofibromatosis type-I (NF1)) (gene has been localised to 17q22). Interestingly, bilateral and symmetrical anteromedial bowing of the tibiae and fibulae has not been described in patients with NF-I.

A benign form of congenital anterolateral bowing of the tibia associated with ipsilateral polydactyly of the hallux: Case report and literature review

Congenital anterolateral bowing of the tibia is generally considered a precursor of congenital pseudarthrosis of the tibia (CPT), which is usually associated with neurofibromatosis type 1 (NF1), a common autosomal dominant genetic disorder. In many cases, NF1 is initially suspected following the presentation of a child with anterolateral tibial bowing. The prognosis of CPT is poor, and amputation may be required. Congenital anterolateral bowing of the tibia combined with ipsilateral polydactyly of the hallux (CABTP) is a rare entity that resembles the anterolateral tibial bowing that occurs in association with CPT, and may be misdiagnosed as NF1. However, spontaneous correction of the tibial bowing with an almost normal fibula has been described in all previously reported cases. Here, we report three patients with CABTP and discuss the physical and imaging characteristics and follow-up results. We suggest that given the spontaneous resolution of bowing, the absence of neurocutaneous signs and the relatively favorable prognosis, CABTP is a distinct entity that merits its own place within the field of anterolateral bowing of the tibia and has no association with CPT or NF1. This should help avoid unnecessary investigations and interventions for NF1. This article shows for the first time tibial duplication in the area of bowing, with two medullary canals surrounded by well-defined cortex on CT.

Congenital anterolateral bowing of the tibia with ipsilateral polydactyly of the great toe associated with cerebral cyst: a new entity?

We present two patients with congenital anterolateral bowing of the tibia with polydactyly who had, in addition, cerebral malformations including agenesis of the corpus callosum and a large cerebral cyst. We discuss phenotypic overlap with the acrocallosal syndrome.

Prophylactic Bypass Grafting and Long-Term Bracing in the Management of Anterolateral Bowing of the Tibia and Neurofibromatosis-1

Congenital anterolateral bowing of the tibia is a pre-pseudarthrosis stage of congenital pseudarthrosis of the tibia and is associated with a substantial risk of fracture and pseudarthrosis. We evaluated the results of prophylactic bypass grafting performed in combination with bracing to prevent fracture and pseudarthrosis. This retrospective series included ten patients with pre-pseudarthrosis of the tibia treated, between 1991 and 2002, with prophylactic bypass grafting with an allograft fibula placed posteromedially in a stress-bearing fashion. The average patient age was 2.3 years at the time of diagnosis, 3.6 years at the time of surgery, and ten years at the time of final follow-up. The mean duration of follow-up was seventy-eight months. Brace protection was recommended at the time of diagnosis and was continued after the operation until maturity. Despite the bracing, a low-energy fracture developed and partially healed prior to the bypass grafting in one patient. The remaining nine patients had no fractures prior to the bypass grafting. At the time of final follow-up, the patients were examined clinically and radiographically for the presence of a fracture or pseudarthrosis and for residual deformity, including malalignment, ankle and knee joint abnormalities, and leg length discrepancy. No patient had either a fracture or a pseudarthrosis of the tibia at the time of follow-up. All grafts united to the tibia at both ends. Complications included an allograft fracture in three patients, which healed in all; allograft resorption in one patient; and pseudarthrosis of the ipsilateral host fibula in one patient. At the time of final follow-up, a mean of 9 mm (range, 0 to 37 mm) of leg length discrepancy was found. A corrective osteotomy was done for four patients (three because of ankle valgus and one because of diaphyseal deformity). In this small series of ten patients with congenital anterolateral bowing of the tibia treated with bypass strut grafting and long-term bracing, there were no cases of pseudarthrosis of the tibia. Distal tibial deformity may persist throughout the growth period and require operative correction in patients treated with this procedure. Therapeutic Level IV. See Instructions to Authors for a complete description of levels of evidence.

Congenital anterolateral tibial bowing and polydactyly: a case report

Congenital anterolateral bowing of the tibia is a rare deformity that may lead to pseudarthrosis and risk of fracture. This is commonly associated with neurofibromatosis type 1. In this report, we describe a 15-month old male with congenital anterolateral bowing of the right tibia and associated hallux duplication. This is a distinct entity with a generally favourable prognosis that should not be confused with other conditions such as neurofibromatosis type 1. Previously published cases are reviewed.

Pathomorphology and treatment of congenital anterolateral bowing of the tibia associated with duplication of the hallux

Congenital unilateral anterolateral tibial bowing in combination with a bifid ipsilateral great toe is a very rare deformity which resembles the anterolateral tibial bowing that occurs in association with congenital pseudarthrosis of the tibia. However, spontaneous resolution of the deformity without operative treatment and with a continuously straight fibula has been described in all previously reported cases. We report three additional cases and discuss the options for treatment. We suggest that this is a specific entity within the field of anterolateral bowing of the tibia and conclude that it has a much better prognosis than congenital pseudarthrosis of the tibia, although conservative treatment alone may not be sufficient.

Anterolateral Tibial Bowing and Duplication of the Hallux: A Rare But Distinct Entity with Good Prognosis

Congenital anterolateral bowing of the lower leg usually is a symptom of (impending) fracture because of congenital pseudarthrosis. This article reports a case of anterolateral bowing of the lower leg that is characterized by spontaneous correction and combination with duplication of the hallux. A review of the literature showed 10 comparable cases. This article is the first report on the magnetic imaging of this entity and describes its differentiation from congenital pseudarthrosis.

Congenital anterolateral bowing of the tibia with ipsilateral polydactyly of the great toe

We report on two cases of congenital unilateral anterolateral bowing and focal defect of the tibia associated with ipsilateral polydactyly of the great toe. Computed tomographic examination showed an unusual partial cleft of the tibia at the site of bowing. A long follow-up of one patient showed spontaneous resolution of the bowing without progression to pseudoarthrosis. These anomalies should be considered as a new entity related to the tibial developmental field.

Congenital anterolateral bowing of the tibia with ipsilateral polydactyly of the great toe

Congenital anterolateral bowing of the tibia with ipsilateral polydactyly of the great toe

Spontaneous resolution of congenital anterolateral bowing of the tibia.

In a series of 43 patients diagnosed with congenital anterolateral bowing of the tibia or congenital pseudarthrosis of the tibia treated between 1980 and 1992, the deformities corrected themselves spontaneously in five patients who had no other congenital abnormalities or evidence of neurofibromatosis. At an average follow-up of 58 months (average age at follow-up 80 months), the only residual deformity seen was a limb-length discrepancy in patients with unilateral involvement. In patients with subperiosteal callus formation on the posteromedial concavity of this deformity and an uninvolved fibula, no treatment appears necessary, as this deformity will spontaneously resolve.