Abstract
Standard surgical repair of esophageal atresia/tracheoesophageal fistula (EA/TEF) is via a right posterolateral thoracotomy. A recognized complication is the later development of scoliosis. The prevalence and pathogenesis of secondary scoliosis are poorly understood. We, therefore, conducted a systematic review on this topic. All English language articles reporting incidence, outcomes and/or interventions for scoliosis in children after EA repair via thoracotomy were identified. Fourteen relevant articles published between 1969 and 2019 reporting 1338 children were included in the analysis. The aggregate prevalence of scoliosis among 937 children without congenital vertebral anomalies was 13%, but this figure varied widely between studies. Severity of scoliosis was documented in 181 children; eight children had a Cobb angle > 40° and 10 had undergone spinal surgery. The spinal curvature in affected individuals was dominantly or exclusively convex to the left. In conclusion, the reported prevalence of scoliosis varies widely but on average affects about one in eight children after open repair of EA/TEF. Most cases are mild and do not require intervention. It is currently uncertain whether secondary scoliosis is preventable by using meticulous thoracotomy techniques or thoracoscopic repair.
Level of evidence IV.
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References
Nassar N, Leoncini E, Amar E et al (2012) Prevalence of esophageal atresia among 18 international birth defects surveillance programs. Birth Defects Res A Clin Mol Teratol 94(11):893–899. https://doi.org/10.1002/bdra.23067
Beasley SW (2006) Esophageal atresia surgical aspects. In: Stringer MD, Oldham KT, Mouriquand PDE (eds) Pediatric surgery and urology long-term outcomes. Cambridge University Press, London, pp 166–180
Moher D, Liberati A, Tetzlaff J, Altman DG, Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 6(7):e1000097. https://doi.org/10.1371/journal.pmed.1000097
Soliman HA, Faure C, Berube G, Mac-Thiong JM, Barchi S, Parent S (2019) Prevalence and natural history of scoliosis and associated congenital vertebral anomalies in patients operated for esophageal atresia with or without tracheoesophageal fistula. J Pediatr Surg 54(7):1308–1311. https://doi.org/10.1016/j.jpedsurg.2018.08.049
Bastard F, Bonnard A, Rousseau V et al (2018) Thoracic skeletal anomalies following surgical treatment of esophageal atresia. Lessons from a national cohort. J Pediatr Surg 53(4):605–609. https://doi.org/10.1016/j.jpedsurg.2017.07.013
Sistonen SJ, Helenius I, Peltonen J, Sarna S, Rintala RJ, Pakarinen MP (2009) Natural history of spinal anomalies and scoliosis associated with esophageal atresia. Pediatrics 124(6):e1198–e1204. https://doi.org/10.1542/peds.2008-3704
Freeman NV, Walkden J (1969) Previously unreported shoulder deformity following right lateral thoracotomy for esophageal atresia. J Pediatr Surg 4(6):627–636. https://doi.org/10.1016/0022-3468(69)90490-4
Koziarkiewicz M, Taczalska A, Jasinska-Jaskula I, Grochulska-Cerska H, Piaseczna-Piotrowska A (2015) Long-term complications of congenital esophageal atresia, single institution experience. Indian Pediatr 52(6):499–501. https://doi.org/10.1007/s13312-015-0664-4
Wei S, Saran N, Emil S (2017) Musculoskeletal deformities following neonatal thoracotomy: long-term follow-up of an esophageal atresia cohort. J Pediatr Surg 52(12):1898–1903. https://doi.org/10.1016/j.jpedsurg.2017.08.062
Okuyama H, Tazuke Y, Uenoa T et al (2017) Long-term morbidity in adolescents and young adults with surgically treated esophageal atresia. Surg Today 47(7):872–876. https://doi.org/10.1007/s00595-016-1462-x
Burford JM, Dassinger MS, Copeland DR, Keller JE, Smith SD (2011) Repair of esophageal atresia with tracheoesophageal fistula via thoracotomy: a contemporary series. Am J Surg 202(2):203–206. https://doi.org/10.1016/j.amjsurg.2010.09.035
Sistonen SJ, Pakarinen MP, Rintala RJ (2011) Long-term results of esophageal atresia: Helsinki experience and review of literature. Pediatr Surg Int 27(11):1141–1149. https://doi.org/10.1007/s00383-011-2980-7
Mortell AE, Azizkhan RG (2009) Esophageal atresia repair with thoracotomy: the Cincinnati contemporary experience. Semin Pediatr Surg 18(1):12–19. https://doi.org/10.1053/j.sempedsurg.2008.10.003
Somppi E, Tammela O, Ruuska T et al (1998) Outcome of patients operated on for esophageal atresia: 30 years' experience. J Pediatr Surg 33(9):1341–1346. https://doi.org/10.1016/s0022-3468(98)90003-3
Chetcuti P, Myers NA, Phelan PD, Beasley SW, Dickens DR (1989) Chest wall deformity in patients with repaired esophageal atresia. J Pediatr Surg 24(3):244–247. https://doi.org/10.1016/s0022-3468(89)80003-x
Jaureguizar E, Vazquez J, Murcia J, Diez Pardo JA (1985) Morbid musculoskeletal sequelae of thoracotomy for tracheoesophageal fistula. J Pediatr Surg 20(5):511–514. https://doi.org/10.1016/s0022-3468(85)80477-2
Gilsanz V, Boechat IM, Birnberg FA, King JD (1983) Scoliosis after thoracotomy for esophageal atresia. AJR Am J Roentgenol 141(3):457–460. https://doi.org/10.2214/ajr.141.3.457
Durning RP, Scoles PV, Fox OD (1980) Scoliosis after thoracotomy in tracheoesophageal fistula patients. A follow-up study. J Bone Jt Surg Am 62(7):1156–1159
Cobb JR (1948) Outline for the study of scoliosis. Instr Course Lect 5:261–275
van Lennep M, Singendonk MMJ, Dall'Oglio L et al (2019) Oesophageal atresia. Nat Rev Dis Primers 5(1):26. https://doi.org/10.1038/s41572-019-0077-0
Soucy P, Bass J, Evans M (1991) The muscle-sparing thoracotomy in infants and children. J Pediatr Surg 26(11):1323–1325. https://doi.org/10.1016/0022-3468(91)90611-v
Sheehan DD, Grayhack J (2017) Pediatric scoliosis and kyphosis: an overview of diagnosis, management, and surgical treatment. Pediatr Ann 46(12):e472–e480. https://doi.org/10.3928/19382359-20171113-01
Hresko MT (2013) Clinical practice. Idiopathic scoliosis in adolescents. N Engl J Med 368(9):834–841. https://doi.org/10.1056/NEJMcp1209063
Konieczny MR, Senyurt H, Krauspe R (2013) Epidemiology of adolescent idiopathic scoliosis. J Child Orthop 7(1):3–9. https://doi.org/10.1007/s11832-012-0457-4
Kaito T, Shimada M, Ichikawa H et al (2018) Prevalence of and predictive factors for scoliosis after surgery for congenital heart disease in the first year of life. JB JS Open Access 3(1):e0045. https://doi.org/10.2106/JBJS.OA.17.00045
Roclawski M, Sabiniewicz R, Potaz P et al (2009) Scoliosis in patients with aortic coarctation and patent ductus arteriosus: does standard posterolateral thoracotomy play a role in the development of the lateral curve of the spine? Pediatr Cardiol 30(7):941–945. https://doi.org/10.1007/s00246-009-9469-3
Lawal TA, Gosemann JH, Kuebler JF, Gluer S, Ure BM (2009) Thoracoscopy versus thoracotomy improves midterm musculoskeletal status and cosmesis in infants and children. Ann Thorac Surg 87(1):224–228. https://doi.org/10.1016/j.athoracsur.2008.08.069
Ruiz-Iban MA, Burgos J, Aguado HJ et al (2005) Scoliosis after median sternotomy in children with congenital heart disease. Spine (Phila Pa 1976) 30(8):E214–E218. https://doi.org/10.1097/01.brs.0000158959.91925.43
Aydin E, Ozler O, Burns P, Lim FY, Peiro JL (2019) Left congenital diaphragmatic hernia-associated musculoskeletal deformities. Pediatr Surg Int 35(11):1265–1270. https://doi.org/10.1007/s00383-019-04548-4
Bisgard JD (1934) Thoracogenic scoliosis: influence of thoracic disease and thoracic operations on the spine. JAMA Surg 29(3):417–445. https://doi.org/10.1001/archsurg.1934.01180030082006
Langenskiold A, Michelsson JE (1961) Experimental progressive scoliosis in the rabbit. J Bone Jt Surg Br 43:116–120
Rothenberg SS (2013) Thoracoscopic repair of esophageal atresia and tracheoesophageal fistula in neonates, first decade's experience. Dis Esophagus 26(4):359–364. https://doi.org/10.1111/dote.12054
Rothenberg SS, Flake AW (2015) Experience with thoracoscopic repair of long gap esophageal atresia in neonates. J Laparoendosc Adv Surg Tech A 25(11):932–935. https://doi.org/10.1089/lap.2015.0124
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Mishra, P.R., Tinawi, G.K. & Stringer, M.D. Scoliosis after thoracotomy repair of esophageal atresia: a systematic review. Pediatr Surg Int 36, 755–761 (2020). https://doi.org/10.1007/s00383-020-04683-3
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DOI: https://doi.org/10.1007/s00383-020-04683-3