nach oben

Erschienen in:

02.03.2022 | Original Article

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

verfasst von: Moritz Markel, Gabrielle Derraugh, Martin Lacher, Shaikh Iqbal, Robert Balshaw, Suyin A. Lum Min, Richard Keijzer

Erschienen in: Pediatric Surgery International | Ausgabe 5/2022

Einloggen, um Zugang zu erhalten

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.

Nur mit Berechtigung zugänglich

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.2017.09.001CrossRefPubMed

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-zCrossRefPubMed

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.2017.02.004CrossRefPubMed

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/s00383-019-04548-4CrossRefPubMed

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/s00383-020-04683-3CrossRefPubMed

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.2018.08.049CrossRefPubMed

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.2014.01.018CrossRefPubMed

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.2017.07.013CrossRefPubMed

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-0033-1350058CrossRef

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/10.1177/1753466620980267CrossRefPubMedPubMedCentral

11.

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

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.23472CrossRefPubMed

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)90152-9CrossRefPubMed

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.2013.02.005CrossRefPubMed

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.2009.10.028CrossRefPubMed

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.12053CrossRefPubMed

Titel: Musculoskeletal deformities in children with congenital thoracic malformations: a population-based cohort study
verfasst von: Moritz Markel
Gabrielle Derraugh
Martin Lacher
Shaikh Iqbal
Robert Balshaw
Suyin A. Lum Min
Richard Keijzer
Publikationsdatum: 02.03.2022
Verlag: Springer Berlin Heidelberg
Erschienen in: Pediatric Surgery International / Ausgabe 5/2022
Print ISSN: 0179-0358
Elektronische ISSN: 1437-9813
DOI: https://doi.org/10.1007/s00383-022-05101-6

Update Pädiatrie

Bestellen Sie unseren Fach-Newsletter und bleiben Sie gut informiert.

Newsletter bestellen

Live-Webinar "Urologie und Sexualmedizin in der Praxis"

Springer Medizin

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