Thoracogenic Spinal Deformity: A Rare Cause of Early Onset Scoliosis International Congress on Early Onset Scoliosis November 19 & 20, 2015 A. Noelle Larson,

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Thoracogenic Spinal Deformity: A Rare Cause of Early Onset Scoliosis International Congress on Early Onset Scoliosis November 19 & 20, 2015 A. Noelle Larson, MD, Sarah Eby, BS, Tricia St. Hilaire, MPH, Michael Glotzbecker, MD, John Smith, MD, Klane White, Children’s Spine Study Group

Congenital diaphragmatic hernia repair Scoliosis in 13-30% (Russell, 2014; Jancelewicz,2010 ) Scoliosis following chest wall resection Superior to 6 th rib: progression odds ratio 10.6 (Glotzbecker, 2013) Progressive scoliosis; convexity toward resection (Kawakami, 1994) Congenital heart defect repair Curves >20° in 8.5% (Reckles, 1975) Introduction

Evaluate patients with severe scoliosis following surgery for congenital conditions Purpose

Multicenter database; patients prospectively enrolled 41 patients with thoracogenic scoliosis (1.7%) Interventions 14 observed 10 braced 1 definitive fusion 16 treated with VEPTR Methods

DiagnosisNon-operativeVEPTR Previous cardiac surgery55 Tracheoesophageal fistula13 Congenital diaphragmatic hernia53 Spinal cord tumor62 Radiation10 Pulmonary hypoplasia12 Chest wall tumor33 Table: Number of subjects with primary underlying diagnosis; one subject in VEPTR group with pulmonary hypoplasia also had congenital diaphragmatic hernia, the other also had previous cardiac surgery. Results Diagnoses

Non- operative VEPTR Total2316 Males156 Females810 Mean age at initial visit (years)5.5 (3.5)6.6 (4.5) Mean follow-up (years)2.9 (2.4)3.8 (3.1) Table: Values given as mean (standard deviation). Results Demographics

Non- operative VEPTR Cobb angle Initial visit33° (12.7°)65° (20.7°) Follow-up visit26° (14.5°)50° (15.7°) Table 2: Pre- and post-treatment. Values given as mean (standard deviation). Results Curve Magnitude

VEPTR group Brachial plexus palsy (1) Resolved with revision of rib hooks Spine infection (2) Hardware failure (1) Device migration (1) Non-operative group – none Results Complications

AB Figure 1. Scoliosis following tracheoesophageal fistula repair. A) Initial curve; Cobb angle = 72°, 13.2 years old; B) Following treatment with VEPTR; Cobb angle = 49°, 15.8 years old. Results Case Example

Heterogeneous sample In many cases, information lacking regarding index surgery Limitations

Scoliosis can develop following thoracotomy, other pediatric surgical procedures Represents <2% of EOS pts in our registry Need prospective study to determine risk factors for development of scoliosis Future work Develop / implement preventative measures Conclusions

Selected references Glotzbecker, M. P., Gold, M., Puder, M., & Hresko, M. T. (2013). Scoliosis after chest wall resection. Journal of Children's Orthopaedics, 7(4), 301–307. Kawakami, N., Winter, R. B., Lonstein, J. E., & Denis, F. (1994). Scoliosis secondary to rib resection. Journal of Spinal Disorders, 7(6), 522–527. Russell, K. W., Barnhart, D. C., Rollins, M. D., Hedlund, G., & Scaife, E. R. (2014). Journal of Pediatric Surgery. Journal of Pediatric Surgery, 49(6), 886–889. Jancelewicz, T., Vu, L. T., Keller, R. L., Bratton, B., Lee, H., Farmer, D., et al. (2010). Long-term surgical outcomes in congenital diaphragmatic hernia: observations from a single institution. Journal of Pediatric Surgery, 45(1), 155– 160. Reckles, L. N., Peterson, H. A., Weidman, W. H., & Bianco, A. J. (1975). The association of scoliosis and congenital heart defects. The Journal of Bone and Joint Surgery. American Volume, 57(4), 449–455.