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      Thoracogenic Spinal Deformity: A Rare Cause of Early Onset Scoliosis

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          Abstract

          Object:

          Surgery for severe congenital defects such as congenital diaphragmatic hernia, congenital heart defects, and tracheoesophageal disorders are life-saving treatments for many infants. However, the incidence of scoliosis following thoracoabdominal surgery ranges from 8­50%. There is little known about severe scoliosis acquired following surgery in infancy. We sought to evaluate patients who developed severe scoliosis following surgical treatment of congenital conditions.

          Methods:

          A multicenter database of patients with early onset scoliosis was queried to identify patients with a history of thoracogenic or acquired scoliosis. Patients with significant congenital spine deformities were excluded. 41 patients (1.6%) were noted to have thoracogenic scoliosis. Of those, 14 were observed, 10 were casted or braced, 17 underwent treatment with rib-based distraction rods, Shilla, or spine-based growing rod devices. Radiographs, complications, and patient characteristics were reviewed.

          Results:

          Mean age at scoliosis diagnosis for the 41 patients was 6.0 years. Mean time to follow-up was 2.9 years. Mean preoperative coronal Cobb angle in the surgical group was 65° and improved to 47° postoperatively ( p=0.01). Mean Cobb angle for the non-operative group was 31° initially and 32° at follow-up (p=0.44). Among the 17 patients undergoing surgery for scoliosis, there were 13 complications in 7 patients, including a brachial plexus palsy following rib-based distraction rod placement. This resolved with revision of the rib hooks. There were no known complications in the non-operative cohort.

          Conclusions:

          Severe scoliosis can develop following thoracotomy and other pediatric surgical procedures. Work is needed to understand the pathogenesis of scoliosis in this population so as to implement preventative measures.

          Level of Evidence:

          IV, retrospective review of prospectively collected data

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          Author and article information

          Journal
          101223545
          32012
          J Neurosurg Spine
          J Neurosurg Spine
          Journal of neurosurgery. Spine
          1547-5654
          1547-5646
          12 March 2019
          01 December 2018
          01 December 2019
          : 29
          : 6
          : 674-679
          Affiliations
          [1 ]Mayo Medical School, Mayo Graduate School, and the Medical Scientist Training Program, College of Medicine, Mayo Clinic, 200 1 st St SW, Rochester, MN
          [2 ]Children’s Spine Study Group, P.O. Box 397, Valley Forge, PA 19481
          [3 ]Children’s Hospital Boston, 300 Longwood Ave, Hunnewell 2, Boston, MA 02115
          [4 ]Department of Orthopaedic Surgery, University of Utah, 100 North Mario Capecchi Drive, Suite 4550, Salt Lake City, Utah 84113
          [5 ]Department of Pediatric Orthopedics, Seattle Children’s Hospital, 4800 Sand Point Way NE, Seattle, WA 98105
          [6 ]Department of Orthopedic Surgery, Mayo Clinic, 200 1 st St SW, Rochester, MN
          Author notes

          AUTHORSHIP:

          Authors include Sarah F. Eby, BS; Tricia St. Hilaire, MPH; Michael Glotzbecker, MD; John Smith, MD; Klane White, MD; Children’s Spine Study Group, and A. Noelle Larson. Data for the project came from the Children’s Spine Study Group registry, which is a multicenter prospective database evaluating patients with early onset scoliosis. Sarah F. Eby, Tricia St. Hilaire, Michael Glotzbecker, John Smith, Klane White, and A. Noelle Larson participated in acquisition, analysis, and interpretation of the data, as well as manuscript preparation and final approval. Sarah Eby and Tricia St. Hilaire performed data abstraction and radiographic review. Sarah Eby wrote the first draft of the manuscript and A. Noelle Larson revised it.

          All authors have participated in the ICMJE criteria:

          •Substantial contributions to the conception or design of the work; or the acquisition, analysis, or interpretation of data for the work; and

          •Drafting the work or revising it critically for important intellectual content; and

          •Final approval of the version to be published; and

          •Agreement to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

          CORRESPONDING AUTHOR: A. Noelle Larson, MD, 200 First St. SW, Rochester, MN 55905, Voice: (507) 284-3660; Fax: (507) 284-5539, Larson.Noelle@ 123456mayo.edu
          Article
          PMC6431290 PMC6431290 6431290 nihpa1012207
          10.3171/2018.4.SPINE171389
          6431290
          30192221
          e1e3bd78-499e-45ed-9623-16172bcd9db0
          History
          Categories
          Article

          VEPTR,infantile,early onset,thoracic wall resection,sternotomy,laminectomy,thoracotomy,scoliosis,diaphragmatic hernia,early onset scoliosis,acquired scoliosis

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