Osteoporosis affects > 1.3 million Australians. Bisphosphonates and denosumab are approved treatments for osteoporosis with the rare complications of jaw osteonecrosis and AFFs. Cases of AFFs were first reported in 2005, highlighting a state of suppressed bone turnover on biopsy (1). In 2010 and 2013, an American Society of Bone and Mineral Research (ASBMR) Task Force proposed a case definition for these atypical fractures affecting the femoral diaphysis (2, 3). Subsequently, reports of similar atypical fractures at other skeletal sites have been published.
Aim
We aimed to systemically identify cases of atypical fractures, excluded from the ASBMR AFF case definition in patients receiving anti-resorptive medication (duration > 3 years).
Method
A structured search of electronic databases (PubMed, Medline, Embase, Cochrane, Web of Science) and hand-searching of conference abstracts/reference lists was completed. All full-text articles written in English describing atypical fractures were screened for: 1) cases of atypical fractures, excluded from the ASBMR AFF case definition in patients (aged > 18 years) receiving long-term antiresorptives, 2) cases published 2005 – 2023.
Results
7954 citations were identified. 65 articles fulfilled the inclusion criteria. Fractures were more common in females (112/120, 93%). Most frequent fracture sites included the ulna (n=32), tibia (n=12), pelvis (n=10), vertebral pedicle (n=8), sacrum (n=6) and femoral neck (n=5). One atypical fracture was reported in a monogenetic bone disorder (hypophosphatasia). Fractures were commonly atraumatic, with prodromal pain and typically transverse, non-comminuted with evidence of cortical thickening and sclerosis. Non-union was more frequent following conservative management. In most cases, anti-resorptive medication was ceased (41/47, 87%).
Conclusion
Atypical fractures at sites other than the femoral diaphysis in patients receiving long-term anti-resorptive treatment are important to recognise and may provide insights into the pathogenesis of AFF. A review of the current AFF case definition could be undertaken to include other skeletal sites.