Journal
GENETICS IN MEDICINE
Volume 18, Issue 6, Pages 570-576Publisher
NATURE PUBLISHING GROUP
DOI: 10.1038/gim.2015.131
Keywords
at-birth fracture; cesarean delivery; in utero fracture; natural history study; osteogenesis imperfecta
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Funding
- Brittle Bone Disease Consortium, a part of the National Center for Advancing Translational Sciences (NCATS) Rare Disease Clinical Research Network [1U54AR068069-0]
- Office of Rare Diseases Research
- NCATS
- National Institute of Arthritis and Musculoskeletal and Skin Diseases (NIAMS)
- National Institute of Dental and Craniofacial Research
- Osteogenesis Imperfecta Foundation
- Baylor College of Medicine Intellectual and Developmental Disabilities Research Center from the Eunice Kennedy Shriver National Institute of Child Health & Human Development [1 U54 HD083092]
- Texas Department of Health Services Summer Scholarship program
- NIH [T32GM07526]
- Doris Duke Charitable Foundation Clinical Scientist Development Award
- Doris Duke Charitable Foundation [2013095]
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Purpose: Osteogenesis imperfecta (OI) predisposes to recurrent fractures. Patients with the moderate to severe forms of OI present with antenatal fractures, and the mode of delivery that would be safest for the fetus is not known. Methods: We conducted systematic analyses of the largest cohort of individuals with OI (n = 540) enrolled to date in the CM Linked Clinical Research Centers. Self-reported at-birth fracture rates were compared among individuals with OI types I, III, and IV. Multivariate analyses utilizing backward-elimination logistic regression model building were performed to assess the effect of multiple covariates, including method of delivery, on fracture-related Outcomes. Results: When accounting for other covariates, at-birth fracture rates did not differ based on whether delivery was by vaginal route or by cesarean delivery (CD). Increased birth weight conferred higher risk for fractures irrespective of the delivery method. In utero fracture, maternal history of OI, and breech presentation were strong predictors for choosing CD. Conclusion: Our study, the largest to analyze the effect of various factors on at-birth fracture rates in OI, shows that CD is not associated with decreased fracture rate. With the limitation that the fracture data were self-reported in this cohort, these results suggest that CD should be performed only for other maternal or fetal indications, not for the sole purpose of fracture prevention in OI.
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