The paradox of prevention—bilateral atypical subtrochanteric fractures due to bisphosphonates in osteogenesis imperfecta

KN Manolopoulos, A West… - The Journal of Clinical …, 2013 - academic.oup.com
The Journal of Clinical Endocrinology & Metabolism, 2013academic.oup.com
A64-year-old woman with osteogenesis imperfecta type 4 was commenced on iv
bisphosphonates after a low-trauma fracture of her ankle. After having received pamidronate
for 3 years and zoledronic acid for 2 years, she complained of a 10-month history of right
lateral thigh and groin pain. She was able to bear weight with only minimal discomfort, and
there was no limitation of movement. Plain radiographs demonstrated an incomplete
subtrochanteric fracture of the lateral cortex of the right femur (Figure 1A) and a similar …
A64-year-old woman with osteogenesis imperfecta type 4 was commenced on iv bisphosphonates after a low-trauma fracture of her ankle. After having received pamidronate for 3 years and zoledronic acid for 2 years, she complained of a 10-month history of right lateral thigh and groin pain. She was able to bear weight with only minimal discomfort, and there was no limitation of movement. Plain radiographs demonstrated an incomplete subtrochanteric fracture of the lateral cortex of the right femur (Figure 1A) and a similar finding at the mid-diaphysis on the left (Figure 1B). Nuclear medicine bone scanning with single photon emission computed tomography (CT) combined with CT revealed intense uptake in the regions corresponding to those identified on the plain x-rays (Figure 1, C and D). Further bisphosphonate treatment was withheld. Teriparatide, a bone anabolic agent, was commenced to promote fracture healing. Osteogenesis imperfecta is typically associated with frequent fractures in childhood and adolescence, but, with age-associated decline in bone density, patients may enter a second period of heightened fracture risk. Our patient had a genetic predisposition to fractures and was treated with bisphosphonates to ameliorate fracture risk (1, 2). Paradoxically, in this case, such treatment is linked to the evolution of bilateral incomplete subtrochanteric fractures (3, 4). Even in patients with unequivocally increased fracture risk (such as in osteogenesis imperfecta), close clinical scrutiny and follow-up are required when using iv bisphosphonates to provide individualized treatment. Careful and sequential reassessments of risks and benefits must be made when managing all patients with bisphosphonates.
Oxford University Press