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Musculoskeletal deformities in children with congenital thoracic malformations: a population-based cohort study

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Abstract

Purpose

It is unclear if musculoskeletal deformities observed in patients with congenital diaphragmatic hernia (CDH), congenital lung lesion (CLL) and esophageal atresia/tracheoesophageal fistula (EA/TEF) are associated with the anomaly or are a result of the surgery required to treat the anomaly. This study compared the prevalence of musculoskeletal deformities for: (1) children with congenital thoracic anomalies to controls; (2) CLL to EA/TEF both repaired via thoracotomy; and (3) CLL and EA/TEF to CDH repaired via laparotomy.

Methods

We performed a retrospective study of children with CLL, CDH or EA/TEF between 1990 and 2016. Date-of-birth-matched control groups were generated from a population-based dataset. International Classification of Disease codes were used to identify scoliosis and pectus anomalies. We determined Hazard ratios (HR) for cases versus controls.

Results

We included 167 cases (CDH n = 82; CLL n = 29; EA/TEF n = 56) and 1670 controls. EA/TEF had a greater risk of scoliosis (HR 5.52, 95%CI 1.49,13.73) and pectus deformities (HR 4.07, 95%CI 1.96,8.45). CDH showed more scoliosis (HR 5.03, 95%CI 1.99,12.74) but not pectus anomalies. Musculoskeletal deformities were not more common in CLL.

Conclusion

Children born with CDH or EA/TEF, but not CLL, had more musculoskeletal deformities than controls. The inconsistent association between musculoskeletal deformities and the surgical approach suggested a congenital predisposition.

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References

  1. Hall NJ, Stanton MP (2017) Long-term outcomes of congenital lung malformations. Semin Pediatr Surg 26:311–316. https://doi.org/10.1053/j.sempedsurg.2017.09.001

    Article  PubMed  Google Scholar 

  2. Kirby E, Keijzer R (2020) Congenital diaphragmatic hernia: current management strategies from antenatal diagnosis to long-term follow-up. Pediatr Surg Int 36:415–429. https://doi.org/10.1007/s00383-020-04625-z

    Article  PubMed  Google Scholar 

  3. van der Zee DC, Tytgat SHA, van Herwaarden MYA (2017) Esophageal atresia and tracheo-esophageal fistula. Semin Pediatr Surg 26:67–71. https://doi.org/10.1053/j.sempedsurg.2017.02.004

    Article  PubMed  Google Scholar 

  4. Aydın E, Özler O, Burns P, Lim FY, Peiró JL (2019) Left congenital diaphragmatic hernia-associated musculoskeletal deformities. Pediatr Surg Int 35:1265–1270. https://doi.org/10.1007/s00383-019-04548-4

    Article  PubMed  Google Scholar 

  5. Mishra PR, Tinawi GK, Stringer MD (2020) Scoliosis after thoracotomy repair of esophageal atresia: a systematic review. Pediatr Surg Int 36:755–761. https://doi.org/10.1007/s00383-020-04683-3

    Article  PubMed  Google Scholar 

  6. Soliman HA, Faure C, Berubé 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 54:1308–1311. https://doi.org/10.1016/j.jpedsurg.2018.08.049

    Article  PubMed  Google Scholar 

  7. Russell KW, Barnhart DC, Rollins MD, Hedlund G, Scaife ER (2014) Musculoskeletal deformities following repair of large congenital diaphragmatic hernias. J Pediatr Surg 49:886–889. https://doi.org/10.1016/j.jpedsurg.2014.01.018

    Article  PubMed  Google Scholar 

  8. Bastard F, Bonnard A, Rousseau V, Gelas T, Michaud L, Irtan S et al (2018) Thoracic skeletal anomalies following surgical treatment of esophageal atresia. Lessons from a national cohort. J Pediatr Surg 53:605–609. https://doi.org/10.1016/j.jpedsurg.2017.07.013

    Article  PubMed  Google Scholar 

  9. Zani A, Eaton S, Hoellwarth M, Puri P, Tovar J, Fasching G et al (2013) International survey on the management of esophageal atresia. Eur J Pediatr Surg 24:003–008. https://doi.org/10.1055/s-0033-1350058

    Article  Google Scholar 

  10. Xie J, Wu Y, Wu C (2020) Is thoracoscopy superior to thoracotomy in the treatment of congenital lung malformations? An updated meta-analysis. Ther Adv Respir Dis 14:1753466620980267. https://doi.org/10.1177/1753466620980267

    Article  PubMed  PubMed Central  Google Scholar 

  11. Drevin G, Andersson B, Svensson JF (2020) Thoracoscopy or Thoracotomy for Esophageal Atresia. Ann Surg. https://doi.org/10.1097/sla.0000000000004239

    Article  Google Scholar 

  12. Concept: Socioeconomic Factor Index ( SEFI ) - Version 2 ( SEFI- 2021;2:2021

  13. Calzolari F, Braguglia A, Valfrè L, Dotta A, Bagolan P, Morini F (2016) Outcome of infants operated on for congenital pulmonary malformations. Pediatr Pulmonol 51:1367–1372. https://doi.org/10.1002/ppul.23472

    Article  PubMed  Google Scholar 

  14. Vanamo K, Peltonen J, Rintala R, Lindahl H, Jääskeläinen J, Louhimo I (1996) Chest wall and spinal deformities in adults with congenital diaphragmatic defects. J Pediatr Surg 31:851–854. https://doi.org/10.1016/S0022-3468(96)90152-9

    Article  CAS  PubMed  Google Scholar 

  15. Jancelewicz T, Chiang M, Oliveira C, Chiu PP (2013) Late surgical outcomes among congenital diaphragmatic hernia (CDH) patients: Why long-term follow-up with surgeons is recommended. J Pediatr Surg 48:935–941. https://doi.org/10.1016/j.jpedsurg.2013.02.005

    Article  PubMed  Google Scholar 

  16. Jancelewicz T, Vu LT, Keller RL, Bratton B, Lee H, Farmer D et al (2010) Long-term surgical outcomes in congenital diaphragmatic hernia: observations from a single institution. J Pediatr Surg 45:155–160. https://doi.org/10.1016/j.jpedsurg.2009.10.028

    Article  PubMed  Google Scholar 

  17. Laberge JM, Blair GK (2013) Thoracotomy for repair of esophageal atresia: Not as bad as they want you to think! Dis Esophagus 26:365–371. https://doi.org/10.1111/dote.12053

    Article  PubMed  Google Scholar 

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Acknowledgements

The authors acknowledge the generosity of the patients and pediatric surgeons of the Winnipeg Health Sciences Center whose experiences made this study possible. This work was supported by the DEVOTION Network and the Children’s Hospital Research Institute of Manitoba. Moritz Markel is holder of a scholarship (MA 8982/1-1) by “Deutsche Forschungsgemeinschaft e.V.” (DFG—German Research Foundation). Dr. Richard Keijzer is the Thorlakson Chair in Surgical Research for the Department of Surgery and the University of Manitoba. The authors acknowledge the Manitoba Centre for Health Policy for use of data contained in the Manitoba Population Research Data Repository under project # 2017/2018-29. The results and conclusions are those of the authors and no official endorsement by the Manitoba Centre for Health Policy, Manitoba Health, or other data providers ins intended or should be inferred. Data used in this study are from the Manitoba Population Research Data Repository housed at the Manitoba Centre for Health Policy, University of Manitoba and were derived from data provided by Manitoba Health. The output of data used for this paper was generated using SAS software, Version 9.4 of the SAS system for Unix. Copyright © 2016 SAS Institute Inc. SAS and all other SAS Institute Inc. product or service names are registered trademarks or trademarks of SAS Institute Inc., Car, NC, USA.

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Authors and Affiliations

  1. Department of Surgery, University of Manitoba, Division of Pediatric Surgery and Children’s Hospital Research Institute of Manitoba, AE402-820 Sherbrook Street, Winnipeg, MB, R3A 1S1, Canada

    Moritz Markel, Suyin A. Lum Min & Richard Keijzer

  2. Department of Pediatric Surgery, University of Leipzig, Leipzig, Germany

    Moritz Markel & Martin Lacher

  3. Master of Physician Assistant Studies, University of Manitoba, Winnipeg, MB, Canada

    Gabrielle Derraugh & Shaikh Iqbal

  4. Department of Pediatrics and Child Health, Division of Respirology, University of Manitoba, Winnipeg, MB, Canada

    Robert Balshaw

  5. Centre for Healthcare Innovation, University of Manitoba, Winnipeg, MB, Canada

    Robert Balshaw

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  1. Moritz Markel
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  2. Gabrielle Derraugh
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  3. Martin Lacher
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  4. Shaikh Iqbal
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  5. Robert Balshaw
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  6. Suyin A. Lum Min
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  7. Richard Keijzer
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Correspondence to Richard Keijzer.

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Markel, M., Derraugh, G., Lacher, M. et al. Musculoskeletal deformities in children with congenital thoracic malformations: a population-based cohort study. Pediatr Surg Int 38, 731–736 (2022). https://doi.org/10.1007/s00383-022-05101-6

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  • DOI: https://doi.org/10.1007/s00383-022-05101-6

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