Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta

S Bellur¹, M Jain¹, D Cuthbertson², D Krakow^{3

4

5}, J R Shapiro⁶, R D Steiner^{7

8

9}, P A Smith¹⁰, M B Bober¹¹, T Hart¹², J Krischer², M Mullins¹, P H Byers^{13

14}, M Pepin^{13

14}, M Durigova¹⁵, F H Glorieux¹⁵, F Rauch¹⁵, V R Sutton^{1

16}, B Lee^{1

16}; Members of the BBD Consortium; S C Nagamani^{1

16}

Affiliations

¹ Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
² College of Medicine, University of South Florida, Tampa, Florida, USA.
³ Department of Orthopedic Surgery, University of California, Los Angeles, California, USA.
⁴ Department of Human Genetics, University of California, Los Angeles, California, USA.
⁵ Department of Obstetrics and Gynecology, University of California, Los Angeles, California, USA.
⁶ Department of Bone and Osteogenesis Imperfecta, Kennedy Krieger Institute, Baltimore, Maryland, USA.
⁷ Department of Pediatrics, Oregon Health & Science University, Portland, Oregon, USA.
⁸ Department of Molecular and Medical Genetics, Oregon Health & Science University, Portland, Oregon, USA.
⁹ Marshfield Clinic Research Foundation and University of Wisconsin, Marshfield and Madison, Wisconsin, USA.
¹⁰ Shriners Hospitals for Children, Chicago, Illinois, USA.
¹¹ Division of Medical Genetics, Alfred I. DuPont Hospital for Children, Wilmington, Delaware, USA.
¹² Osteogenesis Imperfecta Foundation, Gaithersburg, Maryland, USA.
¹³ Department of Medicine, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.
¹⁴ Department of Pathology, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.
¹⁵ Department of Orthopedic Surgery, Shriners Hospital for Children and McGill University, Montreal, Québec, Canada.
¹⁶ Texas Children's Hospital, Houston, Texas, USA.

PMID: 26426884
PMCID: PMC4818203
DOI: 10.1038/gim.2015.131

Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta

S Bellur et al. Genet Med. 2016 Jun.

. 2016 Jun;18(6):570-6.

doi: 10.1038/gim.2015.131. Epub 2015 Oct 1.

Authors

Affiliations

¹ Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.
² College of Medicine, University of South Florida, Tampa, Florida, USA.
³ Department of Orthopedic Surgery, University of California, Los Angeles, California, USA.
⁴ Department of Human Genetics, University of California, Los Angeles, California, USA.
⁵ Department of Obstetrics and Gynecology, University of California, Los Angeles, California, USA.
⁶ Department of Bone and Osteogenesis Imperfecta, Kennedy Krieger Institute, Baltimore, Maryland, USA.
⁷ Department of Pediatrics, Oregon Health & Science University, Portland, Oregon, USA.
⁸ Department of Molecular and Medical Genetics, Oregon Health & Science University, Portland, Oregon, USA.
⁹ Marshfield Clinic Research Foundation and University of Wisconsin, Marshfield and Madison, Wisconsin, USA.
¹⁰ Shriners Hospitals for Children, Chicago, Illinois, USA.
¹¹ Division of Medical Genetics, Alfred I. DuPont Hospital for Children, Wilmington, Delaware, USA.
¹² Osteogenesis Imperfecta Foundation, Gaithersburg, Maryland, USA.
¹³ Department of Medicine, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.
¹⁴ Department of Pathology, Division of Medical Genetics, University of Washington, Seattle, Washington, USA.
¹⁵ Department of Orthopedic Surgery, Shriners Hospital for Children and McGill University, Montreal, Québec, Canada.
¹⁶ Texas Children's Hospital, Houston, Texas, USA.

PMID: 26426884
PMCID: PMC4818203
DOI: 10.1038/gim.2015.131

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.Genet Med 18 6, 570-576.

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Figures

**Figure 1. Birth weight and length of subjects with OI**
Box plots of the birth weight and length in subjects with OI depicting the median, 25th and 75th centiles. The error bars represent the 5th and 95^th centiles. (Pre-term – gestational age < 37 weeks, term gestation - 37–42 weeks; *p<0.01; **p<0.001, Mann-Whitney U Test).

See this image and copyright information in PMC

References

1. Rauch F, Glorieux FH. Osteogenesis imperfecta. Lancet. 2004;363:1377–85. - PubMed
1. Marini JC, Blissett AR. New genes in bone development: what’s new in osteogenesis imperfecta. J Clin Endocrinol Metab. 2013;98:3095–103. - PMC - PubMed
1. Patel RM, et al. A cross-sectional multicenter study of osteogenesis imperfecta in North America - results from the linked clinical research centers. Clin Genet. 2014 - PMC - PubMed
1. Sillence DO, Senn A, Danks DM. Genetic heterogeneity in osteogenesis imperfecta. J Med Genet. 1979;16:101–16. - PMC - PubMed
1. Aerts M, Van Holsbeke C, de Ravel T, Devlieger R. Prenatal diagnosis of type II osteogenesis imperfecta, describing a new mutation in the COL1A1 gene. Prenat Diagn. 2006;26:394. - PubMed

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Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta

Affiliations

Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta

Authors

Affiliations

Abstract

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References

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MeSH terms

Grants and funding

LinkOut - more resources

Full Text Sources

Other Literature Sources

Medical