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Neuraxial and regional anesthesia in surgical patients with osteogenesis imperfecta: a narrative review of literature
  1. Amy R Beethe1,2,
  2. Nicholas A Bohannon3,
  3. Oluwaseye Ayoola Ogun4,
  4. Maegen J Wallace5,6,
  5. Paul W Esposito5,6,
  6. Thomas J Lockhart1,2,
  7. Ryan J Hamlin1,2,
  8. Jasper R Williams1,2 and
  9. Jessica K Goeller1,2
  1. 1 Department of Anesthesiology, University of Nebraska Medical Center, Omaha, Nebraska, USA
  2. 2 Division of Pediatric Anesthesiology, Children's Hospital and Medical Center, Omaha, Nebraska, USA
  3. 3 College of Medicine, University of Nebraska Medical Center, Omaha, Nebraska, USA
  4. 4 Department of Otolaryngology & Probe Analytics Research Institute, LLC, University of Nebraska Medical Center, Omaha, Nebraska, USA
  5. 5 Department of Orthopedic Surgery, University of Nebraska Medical Center, Omaha, Nebraska, USA
  6. 6 Division of Pediatric Orthopedic Surgery, Children's Hospital and Medical Center, Omaha, Nebraska, USA
  1. Correspondence to Dr Amy R Beethe, Children's Hospital and Medical Center, Omaha, NE 68114-4113, USA; abeethe{at}


Background and objectives Regional and neuraxial anesthesia techniques have become instrumental in the perioperative period yet have not been well described in patients with osteogenesis imperfecta (OI), a congenital connective tissue disorder characterized by skeletal dysplasia and fragility. Patients with skeletal dysplasia present unique perioperative challenges that warrant consideration of these techniques despite their relative contraindication in this population due to reports of increased bleeding with surgery, skeletal fragility concerns with positioning, and risk of spinal cord injury with continuous neuraxial catheters. The aim of this narrative review was to evaluate literature describing the use of regional and neuraxial techniques in patients with OI and any associated clinical outcomes.

Methods All available literature from inception to July 2020 was retrieved, according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines, from MEDLINE, Embase, Google Scholar and The Cochrane Library. Three authors reviewed all references for eligibility, abstracted data, and appraised quality.

Results Of 412 articles initially identified, 42 met our inclusion criteria, yielding 161 cases with regional and/or neuraxial techniques described. In 117 (72.6%) of the 161 cases, neuraxial technique was performed, including 76 (64.9%) epidural, 7 (5.9%) caudal, 5 (4.2%) combined spinal epidural, and 29 (24.7%) spinal procedures. In 44 (27.4%) of the 161 cases, the use of regional anesthesia was described. Our review was confounded by incomplete data reporting and small sample sizes, as most were case reports. There were no randomized controlled trials, and the two single-center retrospective data reviews lacked sufficient data to perform meta-analysis. While complications or negative outcomes related to these techniques were not reported in any of the cases, less than half specifically discuss outcomes beyond placement and immediate postoperative course.

Conclusions There is insufficient evidence to validate or refute the potential risks associated with the use of regional and neuraxial techniques in patients with OI. This review did not uncover any reports of negative sequelae related to the use of these modalities to support relative contraindication in this population; however, further research is needed to adequately assess clinically relevant outcomes such as complications and opioid-sparing effect.

  • analgesia
  • nerve block
  • pain
  • postoperative
  • pain management
  • acute pain

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  • Twitter @NBohannonMD, @GoellerJessica

  • Contributors Only those who made important contributions to the study and are included as authors, and all authors are responsible for the content and have read and approved the manuscript for submission to Regional Anesthesia & Pain Medicine.

  • Funding Supported by institutional funding from Children’s Hospital & Medical Center Department of Orthopedic Surgery via the Barbara and Ronald W. Schaefer Endowed Chair Fund of PE.

  • Competing interests None declared.

  • Patient consent for publication Not required.

  • Ethics approval The study was reviewed by the institutional review board and was exempt given its retrospective review of literature nature.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Data availability statement Data sharing not applicable as no datasets generated and/or analyzed for this study. As a narrative review of literature, data only consists of published literature.

  • Author note The manuscript submitted is original, with no portion under simultaneous consideration for publication elsewhere, and has not been previously published.