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Scoliosis in osteogenesis imperfecta: results of posterior spinal fusion in 39 patients

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Abstract

Purpose

To evaluate the outcomes of scoliosis corrective surgery in Osteogenesis Imperfecta (OI) patients with primarily pedicles screw fixation in terms of correcting and maintaining the correction of the spinal deformity, and to assess for several peri-operative parameters and complications associated with this surgical treatment.

Methods

Retrospective case series of 39 consecutive patients with OI treated surgically for scoliosis. The surgeries were performed between 2002 and 2020 by three different surgeons. All patients’ medical peri-operative and post-operative charts were evaluated. Radiological assessment was performed by evaluation of the pre-operative, immediate post-operative and last follow-up plain radiographs.

Results

There were 20 females and 19 males included in this review with a mean age of 14 years (range, 6–20 years) at the time of surgery. The median follow-up time was 7.9 years. The mean pre-operative cobb angle (CA) of the major curve was 76.5 degrees (SD ± 18.9), decreasing to 42.6 (SD ± 17.4) in the long-term post-operative follow-up (P < 0.001). A total of 21 adverse events in 16 patients were noted. Only 4 patients required subsequent invasive surgical treatment or prolonged hospital stay. All other patients were treated conservatively with no lasting complication.

Conclusion

Scoliosis surgical correction in OI patients seems to yield acceptable results, with maintained coronal plane surgical correction in the long-term follow-up. Even though a high peri-operative complications rate is observed in this series, there were no long-term sequelae or lasting complications.

Level of Evidence

Level IV, Case series.

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References

  1. Martin E, Shapiro JR (2007) Osteogenesis imperfecta:epidemiology and pathophysiology. Curr Osteoporos Rep 5:91–97. https://doi.org/10.1007/s11914-007-0023-z

    Article  PubMed  Google Scholar 

  2. Watanabe G, Kawaguchi S, Matsuyama T, Yamashita T (2007) Correlation of scoliotic curvature with Z-score bone mineral density and body mass index in patients with osteogenesis imperfecta: Spine 32(17):E488–E494. https://doi.org/10.1097/BRS.0b013e31811ec2d9

    Article  PubMed  Google Scholar 

  3. Janus GJ, Finidori G, Engelbert RH et al (2000) Operative treatment of severe scoliosis in osteogenesis imperfecta: results of 20 patients after halo traction and posterior spondylodesis with instrumentation. Eur Spine J 9:486–491. https://doi.org/10.1007/s005860000165

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  4. Yong-Hing K, MacEwen GD (1982) Scoliosis associated with osteogenesis imperfecta. J Bone Joint Surg Br 64:36–43. https://doi.org/10.1302/0301-620X.64B1.7068718

    Article  CAS  PubMed  Google Scholar 

  5. Pan CH, Ma SC, Wu CT et al (2006) All pedicle screw fixation technique in correcting severe kyphoscoliosis in an osteogenesis imperfecta patient: a case report. J Spinal Disord Tech 19:368–372. https://doi.org/10.1097/01.bsd.0000208253.06706.42

    Article  PubMed  Google Scholar 

  6. Widmann R F, Bitan F D, Javier Laplaza F, Burke S W, DiMaio M F, Schneider R (1999) Spinal deformity, pulmonary compromise, and quality of life in osteogenesis imperfecta: Spine 24(16):1673. https://doi.org/10.1097/00007632-199908150-00008

    Article  CAS  PubMed  Google Scholar 

  7. Benson DR, Newman DC. (1981) The spine and surgical treatment in osteogenesis imperfecta. Clin Orthop Relat Res.147–153.

  8. Wallace MJ, Kruse RW, Shah SA (2017) The Spine in Patients With Osteogenesis Imperfecta. J Am Acad Orthop Surg 25:100–109. https://doi.org/10.5435/JAAOS-D-15-00169

    Article  PubMed  Google Scholar 

  9. Pinheiro B, Zambrano MB, Vanz AP et al (2019) Cyclic pamidronate treatment for osteogenesis imperfecta: report from a Brazilian reference center. Genet Mol Biol 42:252–260. https://doi.org/10.1590/1678-4685-GMB-2018-0097

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  10. Marginean Ot, Tamasanu R C, Mang N, Mozos I, Brad G F (2017) Therapy with pamidronate in children with osteogenesis imperfecta. Drug Design, Develop Therapy Volume 11:2507–2515. https://doi.org/10.2147/DDDT.S141075

    Article  Google Scholar 

  11. Rauch F, Plotkin H, Zeitlin L et al (2003) Bone mass, size, and density in children and adolescents with osteogenesis imperfecta: effect of intravenous pamidronate therapy. J Bone Miner Res 18:610–614. https://doi.org/10.1359/jbmr.2003.18.4.610

    Article  CAS  PubMed  Google Scholar 

  12. Propst-Proctor SL, Bleck EE (1983) Radiographic determination of lordosis and kyphosis in normal and scoliotic children. J Pediatr Orthop 3:344–346. https://doi.org/10.1097/01241398-198307000-00013

    Article  CAS  PubMed  Google Scholar 

  13. Cheung JPY (2020) The importance of sagittal balance in adult scoliosis surgery. Ann Transl Med 8:35. https://doi.org/10.21037/atm.2019.10.19

    Article  PubMed  PubMed Central  Google Scholar 

  14. Topouchian V, Finidori G, Glorion C et al (2004) Résultats des arthrodèses vertébrales postérieures dans les scolioses secondaires à une ostéogenèse imparfaite [Posterior spinal fusion for kypho-scoliosis associated with osteogenesis imperfecta: long-term results]. Rev Chir Orthop Reparatrice Appar Mot 90:525–532. https://doi.org/10.1016/s0035-1040(04)70426-2

    Article  CAS  PubMed  Google Scholar 

  15. Piantoni L, Noel MA, Francheri Wilson IA et al (2017) Surgical Treatment With Pedicle Screws of Scoliosis Associated With Osteogenesis Imperfecta in Children. Spine Deform 5:360–365. https://doi.org/10.1016/j.jspd.2017.03.002

    Article  PubMed  Google Scholar 

  16. Yilmaz G, Hwang S, Oto M et al (2014) Surgical treatment of scoliosis in osteogenesis imperfecta with cement-augmented pedicle screw instrumentation. J Spinal Disord Tech 27:174–180. https://doi.org/10.1097/BSD.0b013e3182624b76

    Article  PubMed  Google Scholar 

  17. Gupta N, Gupta V, Kumar A et al (2014) Dural ectasia Indian J Anaesth 58:199–201. https://doi.org/10.4103/0019-5049.130829

    Article  PubMed  Google Scholar 

  18. Kollmeier BR, Keenaghan M. (2022) Aspiration Risk. [Updated 2021 Sep 28]. In: StatPearls [Internet]. Available from: https://www.ncbi.nlm.nih.gov/books/NBK470169/

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Funding

This study was not funded. The authors have no relevant financial or non-financial interests to disclose. The study protocol was approved by the Shamir Medical Center institutional review board.

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Contributions

OR drafted the initial manuscript, carried out all statistical analyses, and approved the final manuscript as submitted. JO, NS, KA, AE, YA and YS conceptualized and designed the study, reviewed and revised the manuscript, and approved the final manuscript as submitted. OR and AE designed the data collection instruments, carried out the initial analysis, and approved the final manuscript as submitted. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

Corresponding author

Correspondence to Ahmad Essa.

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Rabau, O., Essa, A., Smorgick, Y. et al. Scoliosis in osteogenesis imperfecta: results of posterior spinal fusion in 39 patients. Eur Spine J 32, 1146–1152 (2023). https://doi.org/10.1007/s00586-023-07550-6

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  • DOI: https://doi.org/10.1007/s00586-023-07550-6

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