Skip to main content

Thank you for visiting nature.com. You are using a browser version with limited support for CSS. To obtain the best experience, we recommend you use a more up to date browser (or turn off compatibility mode in Internet Explorer). In the meantime, to ensure continued support, we are displaying the site without styles and JavaScript.

  • Original Article
  • Published:

Pregnancy outcomes in women with osteogenesis imperfecta: a retrospective cohort study

Journal of Perinatology volume 36, pages 828–831 (2016)Cite this article

Subjects

Abstract

Objective:

Osteogenesis imperfecta (OI) is a rare genetic disorder characterized by defects in type I collagen that can pose serious complications during pregnancy. The aim was to evaluate maternal and fetal outcomes in pregnant women with OI.

Study Design:

This was a retrospective cohort study, using the Healthcare Cost and Utilization Project Nationwide Inpatient Sample. We examined the records of pregnant women with OI during the period 2003 to 2011. We evaluated antenatal complications and method of delivery among 295 women with OI, using unconditional logistic regression.

Results:

Of the total 7 287 994 births in our cohort, we encountered 295 deliveries among women with OI. The prevalence was 4 per 1 00 000 deliveries per year over the study period. Births to women with OI were more likely to be complicated by antepartum hemorrhage (odds ratio (OR) 2.01, 95% confidence interval (CI) 1.04 to 3.91), placenta abruption (OR 2.50, 95% CI 1.24 to 5.03), intrauterine growth restriction and small-for-gestational-age infants (OR 2.42, 95% CI 1.42 to 4.14), congenital malformation (OR 7.33, 95% CI 4.20 to 12.78) and preterm birth (OR 2.24, 95% CI 1.63 to 3.06). Seventy-five percent of women with OI delivered by cesarean section, and they had an increased rate of tubal sterilization at delivery (OR 1.67, 95% CI 1.18 to 2.36). No differences in rates of stress fracture and maternal death were found.

Conclusion:

These findings suggest that there are increased risks to both mother and fetus in pregnancies complicated by OI.

This is a preview of subscription content, access via your institution

Access options

Buy this article

  • Purchase on Springer Link
  • Instant access to full article PDF
Buy now

Prices may be subject to local taxes which are calculated during checkout

Figure 1

Similar content being viewed by others

References

  1. van Dijk FS, Cobben JM, Kariminejad A, Maugeri A, Nikkels PG, van Rijn RR et al. Osteogenesis imperfecta: a review with clinical examples. Mol Syndromol 2011; 2 (1): 1–20.

    CAS  PubMed  PubMed Central  Google Scholar 

  2. Orioli IM, Castilla EE, Barbosa-Neto JG . The birth prevalence rates for the skeletal dysplasias. J Med Genet 1986; 23 (4): 328–332.

    Article  CAS  Google Scholar 

  3. Van Dijk FS, Pals G, Van Rijn RR, Nikkels PGJ, Cobben JM . Classification of osteogenesis imperfecta revisited. Eur J Med Genet 2010; 53 (1): 1–5.

    Article  CAS  Google Scholar 

  4. Chen CP, Lin SP, Su YN, Huang JP, Chern SR, Su JW et al. Uncomplicated vaginal delivery in two consecutive pregnancies carried to term in a woman with osteogenesis imperfecta type I and bisphosphonate treatment before conception. Taiwan J Obstet Gynecol 2012; 51 (2): 305–307.

    Article  Google Scholar 

  5. Feng ZY, Chen Q, Shi CY, Wen HW, Ma K, Yang HX . A type IV osteogenesis imperfecta family and pregnancy: a case report and literature review. Chin Med J (Engl) 2012; 125 (7): 1358–1360.

    Google Scholar 

  6. Key TC, Horger EO 3rd . Osteogenesis imperfecta as a complication of pregnancy. Obstet Gynecol 1978; 51: 67–71.

    CAS  PubMed  Google Scholar 

  7. Sharma A, George L, Erskin K . Osteogenesis imperfecta in pregnancy: two case reports and review of literature. Obstet Gynecol Surv 2001; 56 (9): 563–566.

    Article  CAS  Google Scholar 

  8. Yimgang DP, Shapiro JR . Pregnancy outcomes in women with osteogenesis imperfecta. J Matern Fetal Neonatal Med 2016; 29 (14): 2358–2362.

    PubMed  Google Scholar 

  9. Young BK, Gorstein F . Maternal osteogenesis imperfecta. Obstet Gynecol 1968; 31 (4): 461–470.

    CAS  PubMed  Google Scholar 

  10. Krishnamoorthy U, Vausse S, Donnai P . Management of pregnancy complicated by maternal osteogenesis imperfecta. Report of a case with uterine rupture. J Obstet Gynaecol 2002; 22 (3): 316.

    Article  CAS  Google Scholar 

  11. Litos M, Brown . Osteogenesis imperfecta and pregnancy. Eur J Obstet Gynecol Reprod Biol 2008; 136 (1):126–127.

    Article  Google Scholar 

  12. Vitale MG, Matsumoto H, Kessler MW, Hoffmann W, Roye DP Jr . Osteogenesis imperfecta: determining the demographics and the predictors of death from an inpatient population. J Pediatr Orthop 2007; 27 (2): 228–232.

    Article  Google Scholar 

  13. Hathaway WE, Solomons CC, Ott JE . Platelet function and pyrophosphates in osteogenesis imperfecta. Blood 1972; 39 (4): 500–509.

    CAS  PubMed  Google Scholar 

  14. Chan B, Zacharin M . Maternal and infant outcome after pamidronate treatment of polyostotic fibrous dysplasia and osteogenesis imperfecta before conception: a report of four cases. J Clin Endocrinol Metab 2006; 91 (6): 2017–2020.

    Article  CAS  Google Scholar 

  15. Krakow D. Osteogenesis Imperfecta: A Translational Approach to Brittle Bone Disease, 1st edn. Shapiro JR, Byers P, Florieux F, Sponseller P (eds). Elsevier Inc.: London, UK, 2013.

  16. Cozzolino M, Maggio L, Perelli F, Coccia ME . Ultrasonographic early diagnosis of osteogenesis imperfecta type I: implications for pre and post-natal therapy. Arch Gynecol Obstet 2016; 294 (1): 215–216.

    Article  Google Scholar 

  17. Gotherstrom C, Westgren M, Shaw SW, Astrom E, Biswas A, Byers PH et al. Pre- and postnatal transplantation of fetal mesenchymal stem cells in osteogenesis imperfecta: a two-center experience. Stem Cells Transl Med 2014; 3 (2): 255–264.

    Article  CAS  Google Scholar 

  18. Cubert R, Cheng EY, Mack S, Pepin MG, Byers PH . Osteogenesis imperfecta: mode of delivery and neonatal outcome. Obstet Gynecol 2001; 97 (1): 66–69.

    CAS  PubMed  Google Scholar 

  19. Bellur S, Jain M, Cuthbertson D, Krakow D, Shapiro JR, Steiner RD et al. Cesarean delivery is not associated with decreased at-birth fracture rates in osteogenesis imperfecta. Genet Med 2015; 18 (6): 570–576.

    Article  Google Scholar 

  20. Osteogenesis Imperfecta Foundation. Available at http://www.oif.org/site/PageServer?pagename=Pregnancy. Accessed 5 February 2016.

  21. Smith R, Phillips AJ . Osteoporosis during pregnancy and its management. Scand J Rheumatol Suppl 1998; 107: 66–67.

    Article  CAS  Google Scholar 

  22. Vogel TM, Ratner EF, Thomas RC Jr, Chitkara U . Pregnancy complicated by severe osteogenesis imperfecta: a report of two cases. Anesth Analg 2002; 94 (5): 1315–1317, (table of contents).

    Article  Google Scholar 

  23. Rauch F, Glorieux FH . Osteogenesis imperfecta. Lancet 2004; 363 (9418): 1377–1385.

    Article  CAS  Google Scholar 

  24. Cozzolino M, Perelli F, Maggio L, Coccia ME, Quaranta M, Gizzo S et al. Management of osteogenesis imperfecta type I in pregnancy; a review of literature applied to clinical practice. Arch Gynecol Obstet 2016; 293 (6): 1153–1159.

    Article  Google Scholar 

  25. Carlson JW, Harlass FE . Management of osteogenesis imperfecta in pregnancy. A case report. J Reprod Med 1993; 38 (3): 228–232.

    CAS  PubMed  Google Scholar 

  26. Christodoulou S, Freemont AJ, McVey R, Vause S . Prospective comparative case study of uterine collagen in a woman with osteogenesis imperfecta type 1 who had previously ruptured her uterus. J Obstet Gynaecol 2007; 27 (7): 738–739.

    Article  CAS  Google Scholar 

  27. White NJ, Winearls CG, Smith R . Cardiovascular abnormalities in osteogenesis imperfecta. Am Heart J 1983; 106 (6): 1416–1420.

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Obstetrics and Gynecology, Jewish General Hospital, McGill University, Montréal, QC, Canada

    J Ruiter-Ligeti, T Tulandi & H A Abenhaim

  2. Centre for Clinical Epidemiology and Community Studies, Jewish General Hospital, Montreal, QC, Canada

    N Czuzoj-Shulman, A R Spence & H A Abenhaim

Authors
  1. J Ruiter-Ligeti
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. N Czuzoj-Shulman
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. A R Spence
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. T Tulandi
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. H A Abenhaim
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to H A Abenhaim.

Ethics declarations

Competing interests

Dr Tulandi is an advisor for Actavis Inc. The other authors declare no conflict of interest.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Ruiter-Ligeti, J., Czuzoj-Shulman, N., Spence, A. et al. Pregnancy outcomes in women with osteogenesis imperfecta: a retrospective cohort study. J Perinatol 36, 828–831 (2016). https://doi.org/10.1038/jp.2016.111

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1038/jp.2016.111

This article is cited by

Search

Advanced search

Quick links