Atypical fractures of the femur and ulna and complications of fracture healing in a 76-year-old woman with Sjögren's syndrome.

A 76-years-old Caucasian woman was referred for consultation due to spontaneous fractures of the femur and ulna and complications of fracture healing following surgical treatment of the femur. The patient had a medical history of Sjögren's Syndrome under low doses of methotrexate (7.5-10 mg per os weekly) since 1992, and hypertension. She had osteoporosis since 2001, and had been treated with 6 annual doses of iv zolen-dronic acid 4 mg until 2006, followed by teriparatide 20mg s.c. reported acute onset of spontaneous pain at the lower outer third of the right thigh on May 2006. At that time, plain radiographs of the right femur revealed thickening of the lateral cortex at the supracondylar area with a periosteal stress reaction, while bone-scanning with Tc 99m was suggestive of a supracondylar stress fracture (Figure 1A). Seven months later (December 2006), while walking, the patient sustained an oblique, complete, displaced fracture of the right femur at the same area where the stress fracture had previously occurred. She was then treated surgically with open reduction and internal fixation (90º angu-lated plate and sliding screw), but 21 months later there was no radiological sign of fracture healing, and pseudarthrosis was diagnosed (September 2008) (Figure 1B). One year later (October 2009), and while the fracture had still not healed, the patient sustained a new fracture at the supracondylar area of the right femur at the upper limit of the plate fixation and was operated again, this time with intramedullary nailing. Signs of fracture healing appeared 2 months later the patient suffered a new spontaneous displaced fracture of the right ulna, that was treated with open reduction and internal fix-ation and which healed 4 months later (Figure 1D). It is of note that, at that time (June 2010), the patient presented with laboratory tests with evidence of secondary hyperparathyroidism due to deficiency of 25 (OH) vitamin D3. The patient was treated with high doses of vitamin D (cholecalceferol, 2000 IU per day) from June 2010 to October 2010, when fracture healing of both the femur and ulna was confirmed radiologically. Table 1 illustrates times of fractures as well as complications of healing, in association with medication, bone mineral density testing (DXA) and all laboratory tests available. Commentary Long-term treatment with bisphosphonates has been recently associated with " atypical " subtrochanteric and diaphyseal femoral fractures. According to the ASBMR Task Force Report on atypical femoral fractures 1 , …