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Musculoskeletal deformities in children with congenital thoracic malformations: a population-based cohort study
Moritz Markel1,2, Gabrielle Derraugh3, Martin Lacher2
1Department 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.
Insights
Children with congenital diaphragmatic hernia (CDH) and esophageal atresia/tracheoesophageal fistula (EA/TEF) have higher rates of musculoskeletal deformities. Congenital lung lesions (CLL) did not show this association, suggesting a congenital cause.
Area of Science:
- Pediatric Surgery
- Thoracic Surgery
- Congenital Anomalies
Background:
- Musculoskeletal deformities are observed in infants with congenital thoracic anomalies.
- The etiology of these deformities, whether congenital or surgical, remains unclear.
- Congenital diaphragmatic hernia (CDH), congenital lung lesions (CLL), and esophageal atresia/tracheoesophageal fistula (EA/TEF) are common thoracic anomalies.
Purpose of the Study:
- To compare the prevalence of musculoskeletal deformities in children with congenital thoracic anomalies versus controls.
- To investigate if surgical approach (thoracotomy vs. laparotomy) influences the rate of musculoskeletal deformities in CLL, EA/TEF, and CDH.
Main Methods:
- Retrospective study of 167 children diagnosed with CDH, CLL, or EA/TEF between 1990 and 2016.
- Date-of-birth-matched control groups (n=1670) were utilized for comparison.
- International Classification of Disease codes identified scoliosis and pectus anomalies; Hazard Ratios (HR) were calculated.
Main Results:
- Esophageal atresia/tracheoesophageal fistula (EA/TEF) demonstrated a significantly higher risk for scoliosis (HR 5.52) and pectus deformities (HR 4.07).
- Congenital diaphragmatic hernia (CDH) showed an increased risk for scoliosis (HR 5.03) but not pectus anomalies.
- Congenital lung lesions (CLL) did not exhibit a higher prevalence of musculoskeletal deformities compared to controls.
Conclusions:
- Children with CDH and EA/TEF have a greater predisposition to developing musculoskeletal deformities.
- Congenital lung lesions (CLL) are not associated with an increased risk of these deformities.
- The findings suggest a congenital origin for these deformities, independent of the surgical intervention.
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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