title: An atypical fracture in male patient with osteogenesis imperfecta.
abstract: So-called atypical fractures have been related to prolonged treatment with bisphosphonates. Although there remain unanswered questions with respect to their etiology and physiopathology, it does appear to be a causal relationship. There are many references in the literature about this problem in patients in whom these drugs have been used to treat osteoporosis, but few reports in patients who have received this therapy for the management of osteogenesis imperfecta. The Authors describe a case of a young male patient with osteogenesis imperfecta with a number of historical fractures, and who received treatment with these drugs, initially parenterally and subsequently orally, presenting as a complication of the treatment, an atypical diaphyseal femoral fracture. The characteristics of the fracture are consistent with the updated diagnostic criteria of the American Society for Bone and Mineral Research. The clinical case, its treatment, both surgically and metabolically with teriparatide, and its development over a year, are analysed. The case is notable for, on the one hand, the significance of the presence of this type of fracture in a young patient with this disease, and on the other, because of the administration of teriparatide outside its established clinical indications, with twin objectives: to improve the bone structure of the patient's underlying disease, and to counteract the harmful effects which bisphosphonates may have on this bone.
authors.affiliation: Orthopaedic Surgery and Traumatology Department, Alto Deba Hospital, Mondragón, Gipuzkoa, Spain.
authors.forename: Iñigo
authors.initials: I
authors.lastname: Etxebarria-Foronda
authors.affiliation: Orthopaedic Surgery and Traumatology Department, Reina Sofia University Hospital, Córdoba, Spain.
authors.forename: Pedro
authors.initials: P
authors.lastname: Carpintero
keywords: atypical femoral fracture; bisphosphonates; osteogenesis imperfecta; teriparatide
references.citation: J Clin Invest. 2014 Feb;124(2):491-8
references.pmid: 24463451
references.citation: Endocr Pract. 2014 Oct;20(10 ):e187-90
references.pmid: 24936568
references.citation: J Bone Miner Metab. 2016 Mar;34(2):123-31
references.pmid: 26031935
references.citation: Rev Esp Cir Ortop Traumatol. 2015 Nov-Dec;59(6):373-93
references.pmid: 26233814
references.citation: J Bone Miner Res. 2014 Jan;29(1):1-23
references.pmid: 23712442
references.citation: J Pediatr Orthop. 2016 Oct-Nov;36(7):757-61
references.pmid: 26090982
references.citation: J Bone Miner Res. 2009 Dec;24(12):1998-2006
references.pmid: 19453263
references.citation: Clin Exp Rheumatol. 2000 Sep-Oct;18(5):654
references.pmid: 11072619
references.citation: Pediatrics. 2007 Mar;119 Suppl 2:S163-5
references.pmid: 17332237
references.citation: J Bone Miner Res. 2015 May;30(5):929-33
references.pmid: 25407702
references.citation: J Orthop Trauma. 2014 Jan;28(1):57-62
references.pmid: 23454854
references.citation: Osteoporos Int. 2011 Dec;22(12):2951-61
references.pmid: 21997225
references.citation: Joint Bone Spine. 2015 Jul;82(4):287-8
references.pmid: 25543273
references.citation: J Clin Endocrinol Metab. 2013 Mar;98 (3):871-2
references.pmid: 23386638
references.citation: Front Endocrinol (Lausanne). 2013 Aug 26;4:106
references.pmid: 23986744
references.citation: Am J Ther. 2016 May-Jun;23 (3):e894-904
references.pmid: 25844482
references.citation: Calcif Tissue Int. 2013 Nov;93(5):448-52
references.pmid: 23907723
references.citation: Acta Orthop. 2012 Oct;83(5):548-50
references.pmid: 22998530
references.citation: Rev Esp Cir Ortop Traumatol. 2014 Mar-Apr;58(2):114-9
references.pmid: 24438858