Amputation Outcomes in Congenital Pseudarthrosis of the Tibia

Westberry, David E.; Carpenter, Ashley M; Tisch, Jocelyn; Wack, Linda I

doi:10.1097/bpo.0000000000001211

Cited by 16 publications

(12 citation statements)

References 31 publications

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“…Some researchers believe that the involved tibia in patients should be amputated early to preserve extremity function with an adequate prosthesis because the residual deformities impair the affected lower-extremity walking function. 16 One of the typical residual deformities was tibial malalignment in the frontal plane, which includes proximal tibial valgus and ankle valgus deformities and tibial diaphyseal angulation. To address this rather intractable problem, Charnley 17 first reported that using an IMR to transfix the talus and calcaneus successfully treated two cases of CPT in 1956, demonstrating that an IMR can maintain the involved tibial alignment and turn abnormal angulation strains into longitudinal compression forces to reduce the possibility of refracture occurrence.…”

Section: Discussionmentioning

confidence: 99%

The association between fibular status and frontal plane tibial alignment post-union in congenital pseudarthrosis of the tibia

Deng

Mei

Wang

et al. 2021

Journal of Children's Orthopaedics

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Purpose Controversy exists regarding fibular status related to tibial alignment after congenital pseudarthrosis of the tibia (CPT) achieves union. We aimed to determine whether fibular status affected frontal plane tibial alignment post-CPT union. Methods Between January 2010 and December 2013, we followed 36 patients treated using a combined surgical technique (mean 6.6 years (2.2 to 9.4)). Outcome measures comprised medial proximal tibial angle, tibial diaphyseal angulation, lateral distal tibial angle (LDTA), relative intramedullary rod length and relative fibula length discrepancy. Results The frequency of proximal tibial valgus deformity and ankle valgus deformity were significantly lower in the fibular integrity group than in the fibular un-integrity group (proximal tibial valgus deformity: 2/17 versus 11/19; p = 0.006; ankle valgus deformity: 10/17 versus 18/19; p = 0.016). The mean LDTA was notably higher in the fibular integrity group than in the fibular un-integrity group (81.2° (sd 6.7°) versus 71.3° (sd 6.6°); p < 0.001). A statistically significant difference was observed in relative fibula length discrepancy between patients with fibular integrity (0.06 (0 to 0.11); Malhotra grade 0, n = 6; Malhotra grade I, n = 11) and those without (0.24 (0.01 to 0.36); Malhotra grade 2, n = 2; Malhotra grade 3, n = 17; p < 0.001). Ankle valgus deformity had a significant positive correlation with proximal tibial valgus deformity (r = 0.402; p = 0.015). Patients with an intact and normal length fibula had a lower frequency of ankle valgus deformity than those with an intact but shortening fibula (1/6 versus 9/11; p = 0.035). Conclusion Shortening or pseudarthrosis of the fibula was associated with ankle valgus and proximal tibial valgus deformities post-CPT union. Level of evidence III

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Section: Discussionmentioning

confidence: 99%

The association between fibular status and frontal plane tibial alignment post-union in congenital pseudarthrosis of the tibia

Deng

Mei

Wang

et al. 2021

Journal of Children's Orthopaedics

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“…Neurofibromatous osteopathy is one of the most challenging conditions for pediatric orthopedic surgeons, and many operations will be required for successful treatment. [7] Currently, most patients are treated by excising the lesions, stabilizing the fragments with internal or external fixation, and bone grafting. In addition to this basic approach, some surgeons advocate the local administration of bone morphogenetic protein, [8,9] periosteal grafting, [10] and vascularized fibular grafts.…”

Section: Discussionmentioning

confidence: 99%

Unusual form of the distal bone defect of ulna with neurofibromatosis type 1

Shen

Chen

2019

Medicine

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Rationale:Bone malformation occurs in 10% to 25% neurofibromatosis type 1 (NF-1) patients, and the manifestations are scoliosis, congenital arch and pseudo-joint formation, bone cyst, and pathologic fracture. However, a large segmental defect without obvious signs of bone destruction has rarely been reported.Patient concerns:A 4.5-year-old male presented with a 4-year history of shortening of the right upper limb and radial head dislocation. The X-ray indicated a lack of the distal part of the right ulna and radial head dislocation.Diagnosis:The X-ray showed obvious bone resorption at the right ulna distal, proximal stubble, and distal part of the epiphyseal residue, which was 4.3 mm shorter after 14 months. The patient was finally diagnosed with NF-1 according to the pathologic examination.Interventions:The treatment included tumor resection, ulnar osteotomy, and fixation by an Ilizarov frame.Outcomes:The Ilizarov frame was removed after 2.7 months of surgery. The radial head was successfully repositioned, and the elbow joint function was significantly improved. No recurrence of the deformity was noted until now.Lessons:Osteolysis (defect without bone destruction) is an extremely rare symptom in patients with NF1. Therefore, it is essential to make the right diagnosis by comprehensive and careful physical examination.

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“…Neurofibromas that are occasionally extending in large areas of the tibia, is difficult to be surgically removed. In these cases, amputation of the limb and use of prosthesis can be proposed [45,46].…”

Section: Osseous Involvement Of Long Bones 31 Tibiamentioning

confidence: 99%

Bone Lesions in Children with Neurofibromatosis

Laliotis¹

2022

Clinical and Basic Aspects of Neurofibromatosis Type 1

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Neurofibromatosis is often related with severe orthopaedic disorders in children. Bone lesions are rare but pose severe difficulties in management. It affects the spine and long bones. Lesions are associated either from enlargement of neurofibromas that affect the normal growth or from primary neurofibromatosis of long bones. Dystrophic scoliosis appears with short curves, with kyphosis and rotation of the apical vertebrae. Usually affect the thoracic spine, with penciling of the ribs. Surgical treatment is challenging in cases of rapid progression. Scoliosis may appear with curvatures similar to those in idiopathic scoliosis, without dysplastic changes of the vertebrae. Anterior bowing of the tibia is manifestation of NF and is distinguished from the benign posterolateral bowing. Evaluation of the medullary canal and presence of cystic lesions in the tibia is essential. Progression to pseudoarthrosis or pathologic fracture is common. Surgical management of tibial pseudoarthrosis remains a difficult procedure. Pseudoarthrosis may appear in fibula, radius or ulna but are extremely rare. Irregular eccentric bone cysts in long bones that are commonly diagnosed after a pathologic fracture, must be differentiated for NF. Malignant transformation of neurofibromas must be considered when there is rapid progression of the lesion.

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Amputation Outcomes in Congenital Pseudarthrosis of the Tibia

Abstract: Level IV-case series.

Cited by 16 publications

References 31 publications

The association between fibular status and frontal plane tibial alignment post-union in congenital pseudarthrosis of the tibia

The association between fibular status and frontal plane tibial alignment post-union in congenital pseudarthrosis of the tibia

Unusual form of the distal bone defect of ulna with neurofibromatosis type 1

Bone Lesions in Children with Neurofibromatosis

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