@article{6a3c5e5c26284362bfc7a3f946857a9f,
title = "Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta",
abstract = "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 OI 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.",
keywords = "at-birth fracture, cesarean delivery, in utero fracture, natural history study, osteogenesis imperfecta",
author = "S. Bellur and M. Jain and D. Cuthbertson and D. Krakow and Shapiro, {J. R.} and Steiner, {R. D.} and Smith, {P. A.} and Bober, {M. B.} and T. Hart and J. Krischer and M. Mullins and Byers, {P. H.} and M. Pepin and M. Durigova and Glorieux, {F. H.} and F. Rauch and Sutton, {V. R.} and B. Lee and Nagamani, {S. C.}",
note = "Funding Information: This work was supported by the Brittle Bone Disease Consortium (1U54AR068069-0), a part of the National Center for Advancing Translational Sciences (NCATS) Rare Disease Clinical Research Network, and is funded through a collaboration between the Office of Rare Diseases Research, NCATS, the National Institute of Arthritis and Musculoskeletal and Skin Diseases (NIAMS), and the National Institute of Dental and Craniofacial Research. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health (NIH). The Brittle Bone Disease Consortium is also supported by the Osteogenesis Imperfecta Foundation. The project was also supported by Baylor College of Medicine Intellectual and Developmental Disabilities Research Center (grant 1 U54 HD083092) from the Eunice Kennedy Shriver National Institute of Child Health & Human Development. S.B. was the recipient of the Texas Department of Health Services Summer Scholarship program for 2014. M.J. was funded by an NIH training grant (T32GM07526). S.C.N. is a recipient of the Doris Duke Charitable Foundation Clinical Scientist Development Award, and this work was supported by the Doris Duke Charitable Foundation (grant 2013095). The authors thank the members of the Brittle Bone Disease Consortium: David Eyre, Gerald Harris, Eric Orwoll, Cathleen Raggio, Laura Tosi, and Anne Tsai. They also recognize the contributions of clinical research teams at the respective sites: Mary Mullins, Alyssa Tran, Susan Carter (Baylor College of Medicine and Texas Children's Hospital); Vonda Vensel, Jill Christie, and Abigail Hata (Oregon Health & Science University); Michaela Durigova (Shriners Hospital Montreal); and Lauren Davey (A.I. duPont Hospital for Children). Publisher Copyright: {\textcopyright} 2016 American College of Medical Genetics and Genomics.",
year = "2016",
month = jun,
day = "1",
doi = "10.1038/gim.2015.131",
language = "English (US)",
volume = "18",
pages = "570--576",
journal = "Genetics in Medicine",
issn = "1098-3600",
publisher = "Lippincott Williams and Wilkins",
number = "6",
}