Successful treatment of pathologic femoral shaft fracture associated with large arteriovenous malformations using a 3-dimensional external fixator and teriparatide: a case report

Abstract

Background Arteriovenous malformations (AVMs) are rare congenital vascular lesions associated with early quiescence, late expansion, and, ultimately, infiltration and destruction of local soft tissue and bone. The extremities are a common location. Incidence of bony involvement by AVM has been reported as high as 31%. However, there are few reports on management of pathologic fracture associated with AVM. Teriparatide is a recombinant parathyroid hormone (PTH) analogue consisting of the 1–34 fragment of PTH. Recently, some reports have shown the ability of teriparatide to improve fracture healing. Here, we present a case of pathologic femoral shaft fracture associated with large AVMs that was treated successfully by external fixation and teriparatide. Case presentation A 68-year-old Japanese woman, previously diagnosed as having large AVMs, sustained a right femoral shaft fracture due to a fall. At the time of admission, she presented with massive swelling and venous varicosities of the right thigh. Plain radiography of the right thigh revealed femoral shaft fracture with bony erosion and calcification of soft tissue. We planned closed reduction and intramedullary nailing with a unilateral external fixator following embolization of the feeding artery. However, closed reduction using the fracture table was difficult. When we attempted open reduction, massive bleeding (1000 mL) after incision of subcutaneous tissue occurred. Finally, we carefully applied a Taylor Spatial Frame. Fracture displacement was corrected successfully and bony union was obtained with administration of teriparatide 15 months after the initial surgery. The patient is able to walk using 1 cane. Conclusion We present the first report of pathologic fracture associated with large AVMs that achieved bony union using a 3-dimensional external fixator and teriparatide.

Use of teriparatide in preventing delayed bone healing in complex biosseous leg fracture: a case report

“…We opted to use systemic therapy with teriparatide prior to the possible onset of pseudoarthrosis, simultaneously with mechanical stimulation obtained through dynamization, in order to enhance the bone healing, achieve an adequate callus, and prevent delayed bone healing. The positive effect of teriparatide on fracture healing is observed in animals, but several case reports also describe a positive effect in humans, although more studies are necessary to confirm this [6][7][8][9][10][11][12][13][14][15]24] . The effect of teriparatide is due to its capacity to stimulate bone progenitor cells [25,26] .…”

“…Nowadays, the efficacy of teriparatide in treating osteoporosis is widely accepted. Instead, the debate on its use to enhance fracture healing has yet to lead to a consensus, although it has been described as effective in numerous case reports and is a promising treatment to improve the healing of nonunion fractures [9][10][11][12][13][14][15] . In this report, we present the case of a patient with a complex open biosseous fracture of the leg which was addressed through surgery and early systemic treatment with teriparatide 20 mcg/day for 3 months, in order to prevent delayed union of the fracture.…”

Hypoplastic femur with pathologic femoral shaft fracture associated with extensive arteriovenous malformations. A series of five cases

“…AVMs most commonly occur in the extremities, facial bone, and pelvis and are usually goes unnoticed, which leads to late expansion and ultimately, infiltration and destruction of local soft tissue and bone [ 3 ]. AVMs can also lead to local bony changes by weakening them and finally pathologic fracture [ 4 , 5 , 6 ]. There are several treatment options for AVM, including conservative therapy with fitted pressure corticosteroids [ 7 ], garments systemic [ 8 ], embolization [ 9 ], radiation [ 10 , 11 ], sclerotherapy [ 12 ], and surgical removal [ 13 ], or a combined of these modalities [ 14 , 15 ].…”

“…These lesions are commonly seen in extremities, facial bone and pelvis, and usually goes unnoticed, which leads to expansion, infiltration, and ultimately destroy underlying tissue and bone [ 3 ]. AVMs can also lead to local bony changes by causing improper growth and hypoplasia that weakens them and finally leads to pathologic fracture [ 4 , 5 , 6 ]. Many treatment modalities are available for AVM management, including non-surgical therapy like fitted pressure corticosteroids [ 7 ], garments systemic [ 8 ], embolization [ 9 ], radiation [ 10 , 11 ], sclerotherapy [ 12 ], and surgical removal [ 13 ], or a combine approach [ 14 , 15 ].…”

“…The incidence of skeletal involvement especially long bones by AVM has been reported to be 20 to 31% [ 13 , 16 ]. In the existing literature, few similar cases reported on the management of pathologic fracture that coexisted with AVM [ 5 , 17 , 18 ].…”