title: Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta.
abstract: 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.
  
  
  
  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.
  
  
  
  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.
  
  
  
  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.Genet Med 18 6, 570-576.
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
authors.forename: S
authors.initials: S
authors.lastname: Bellur
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
authors.forename: M
authors.initials: M
authors.lastname: Jain
authors.affiliation: College of Medicine, University of South Florida, Tampa, Florida, USA.
authors.forename: D
authors.initials: D
authors.lastname: Cuthbertson
authors.affiliation: Department of Orthopedic Surgery, University of California, Los Angeles, California, USA.|Department of Human Genetics, University of California, Los Angeles, California, USA.|Department of Obstetrics and Gynecology, University of California, Los Angeles, California, USA.
authors.forename: D
authors.initials: D
authors.lastname: Krakow
authors.affiliation: Department of Bone and Osteogenesis Imperfecta, Kennedy Krieger Institute, Baltimore, Maryland, USA.
authors.forename: J R
authors.initials: JR
authors.lastname: Shapiro
authors.affiliation: Department of Pediatrics, Oregon Health & Science University, Portland, Oregon, USA.|Department of Molecular and Medical Genetics, Oregon Health & Science University, Portland, Oregon, USA.|Marshfield Clinic Research Foundation and University of Wisconsin, Marshfield and Madison, Wisconsin, USA.
authors.forename: R D
authors.identifier: 0000-0003-4177-4590
authors.initials: RD
authors.lastname: Steiner
authors.affiliation: Shriners Hospitals for Children, Chicago, Illinois, USA.
authors.forename: P A
authors.initials: PA
authors.lastname: Smith
authors.affiliation: Division of Medical Genetics, Alfred I. DuPont Hospital for Children, Wilmington, Delaware, USA.
authors.forename: M B
authors.identifier: 0000-0002-6178-1264
authors.initials: MB
authors.lastname: Bober
authors.affiliation: Osteogenesis Imperfecta Foundation, Gaithersburg, Maryland, USA.
authors.forename: T
authors.initials: T
authors.lastname: Hart
authors.affiliation: College of Medicine, University of South Florida, Tampa, Florida, USA.
authors.forename: J
authors.initials: J
authors.lastname: Krischer
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
authors.forename: M
authors.initials: M
authors.lastname: Mullins
authors.affiliation: Department of Medicine, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.|Department of Pathology, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.
authors.forename: P H
authors.identifier: 0000-0001-7786-7030
authors.initials: PH
authors.lastname: Byers
authors.affiliation: Department of Medicine, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.|Department of Pathology, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.
authors.forename: M
authors.identifier: 0000-0001-6299-2971
authors.initials: M
authors.lastname: Pepin
authors.affiliation: Department of Orthopedic Surgery, Shriners Hospital for Children and McGill University, Montreal, Québec, Canada.
authors.forename: M
authors.initials: M
authors.lastname: Durigova
authors.affiliation: Department of Orthopedic Surgery, Shriners Hospital for Children and McGill University, Montreal, Québec, Canada.
authors.forename: F H
authors.initials: FH
authors.lastname: Glorieux
authors.affiliation: Department of Orthopedic Surgery, Shriners Hospital for Children and McGill University, Montreal, Québec, Canada.
authors.forename: F
authors.initials: F
authors.lastname: Rauch
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.|Texas Children's Hospital, Houston, Texas, USA.
authors.forename: V R
authors.initials: VR
authors.lastname: Sutton
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.|Texas Children's Hospital, Houston, Texas, USA.
authors.forename: B
authors.initials: B
authors.lastname: Lee
authors.affiliation: Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.|Texas Children's Hospital, Houston, Texas, USA.
authors.forename: S C
authors.initials: SC
authors.lastname: Nagamani
mesh_terms: D001724:Birth Weight / Q000235:genetics; D002585:Cesarean Section / Q000009:adverse effects*; D005260:Female; D050723:Fractures, Bone / Q000175:diagnosis / Q000209:etiology / Q000503:physiopathology*; D006801:Humans; D007231:Infant, Newborn; D016015:Logistic Models; D008297:Male; D010013:Osteogenesis Imperfecta / Q000175:diagnosis / Q000209:etiology / Q000503:physiopathology*; D011247:Pregnancy; D011296:Prenatal Diagnosis