Pediatr Surg Int

. 2022 Mar 2. doi: 10.1007/s00383-022-05101-6. Online ahead of print. https://pubmed.ncbi.nlm.nih.gov/35235016/

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

Moritz Markel ^1 2, Gabrielle Derraugh ³, Martin Lacher ², Shaikh Iqbal ³, Robert Balshaw ^4 5, Suyin A Lum Min ¹, Richard Keijzer ⁶Affiliations expand

• PMID: 35235016
• DOI: 10.1007/s00383-022-05101-6

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.

Keywords: Congenital diaphragmatic hernia; Congenital lung lesion; Esophageal atresia; Pectus carinatum; Pectus excavatum; Scoliosis.

© 2022. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.