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Their occurrence in patients with osteogenesis imperfecta (OI), a hereditary bone disease leading to bone fragility, is not well understood. In this study, 138 adults with genetically confirmed classical OI were screened for AFF. Five patients with AFF were identified and compared to an age- and treatment-matched adult OI cohort without AFF (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u200923). Demographical parameters, biochemical markers, bone mineral density (DXA), bone microarchitecture (HR-pQCT), and radiographs were analyzed. In addition, antiresorptive therapy and the duration of treatment were determined and compared. In the screened OI cohort, AFF prevalence was 3.6%. No significant differences were observed between groups regarding age, weight, height, BMI, fracture history, bone mineral density, or antiresorptive therapy exposure and duration. HR-pQCT showed no significant microarchitectural differences, although a trend toward higher cortical thickness was noted in AFF patients. AFF are a rare complication in adults with classical OI and appear to be multifactorial in origin. Our findings suggest that AFF are not exclusively related to antiresorptive therapy but may be influenced by disease-specific factors, particularly the underlying collagen defect, femoral deformities and altered biomechanics. Individualized management strategies are essential, and further studies are needed to clarify underlying mechanisms and best treatment options.\n                  <\/jats:p>","DOI":"10.1007\/s00223-026-01576-w","type":"journal-article","created":{"date-parts":[[2026,7,18]],"date-time":"2026-07-18T06:22:04Z","timestamp":1784355724000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Atypical Femoral Fractures in Adult Patients with Classical Osteogenesis Imperfecta"],"prefix":"10.1007","volume":"117","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-9094-6021","authenticated-orcid":false,"given":"Mikolaj","family":"Bartosik","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0006-7522-4887","authenticated-orcid":false,"given":"Oskar","family":"Windels","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0200-8431","authenticated-orcid":false,"given":"Florian","family":"Barvencik","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2382-8348","authenticated-orcid":false,"given":"Michael","family":"Amling","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9449-5013","authenticated-orcid":false,"given":"Ralf","family":"Oheim","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,7,18]]},"reference":[{"key":"1576_CR1","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1002\/jbmr.1998","volume":"29","author":"E Shane","year":"2014","unstructured":"Shane E, Burr D, Abrahamsen B, Adler RA, Brown TD, Cheung AM, Cosman F, Curtis JR, Dell R, Dempster DW, Ebeling PR, Einhorn TA, Genant HK, Geusens P, Klaushofer K, Lane JM, McKiernan F, McKinney R, Ng A, Nieves J, O\u2019Keefe R, Papapoulos S, Howe TS, van der Meulen MC, Weinstein RS, Whyte MP (2014) Atypical subtrochanteric and diaphyseal femoral fractures: second report of a task force of the American society for bone and mineral research. 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OW received travel support from Alexion, Inozyme and UCB and served as a speaker for Alexion. RO has served as a speaker and advisory board member for Hexal Pharma, Kyowa Kirin, Inozyme, Ipsen, Mereo, Pharmacosmos and UCB and has received institutional research grants from Kyowa Kirin, UCB and Inozyme. FB has received speaker fees from Alexion, UCB, Blueprint Medicines (Germany), Synlab, Gedeon Richter and Diasorin and has received institutional research grants from UCB and Alexion. MA declares that there is no conflict of interest.","order":1,"name":"Ethics","label":"Conflict of interest","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"This retrospective study was performed in accordance with the local ethical guidelines (PV5364 and 2023-101124-BO-ff) and the Declaration of Helsinki.","order":2,"name":"Ethics","label":"Ethical Approval","group":{"name":"EthicsHeading","label":"Declarations"}}],"article-number":"115"}}