Lung function, exercise tolerance, and physical growth of children with congenital lung malformations at 8 years of
Annelieke Hijkoop1, Marloes M van Schoonhoven1, Joost van Rosmalen2
1Department of Pediatric Surgery and Intensive Care, Erasmus MC - Sophia Children's Hospital, Rotterdam, The Netherlands.
Insights
Children with congenital lung malformations (CLM) may experience reduced lung function and exercise tolerance, particularly after surgery. A watchful waiting approach is supported for asymptomatic CLM cases.
Area of Science:
- Pediatric Pulmonology
- Congenital Lung Malformations
- Long-term Health Outcomes
Background:
- Congenital lung malformations (CLM) require careful management to optimize long-term health.
- Understanding the long-term pulmonary and exercise outcomes in children with CLM is crucial for effective counseling.
- Comparing surgical versus observational management strategies provides insights into disease progression and treatment efficacy.
Purpose of the Study:
- To evaluate and compare the long-term lung function, exercise tolerance, and physical growth in children with CLM based on their management (surgery vs. observation).
- To provide data that can improve counseling for families regarding the expected outcomes of CLM.
- To identify risk factors and long-term consequences associated with different management approaches for CLM.
Main Methods:
- A longitudinal prospective follow-up study involving 61 children diagnosed with CLM.
- Assessment of lung function using spirometry and exercise tolerance via the Bruce treadmill test at 8 years of age.
- Statistical analysis using standard deviation scores (SDS) and general linear models to compare outcomes between surgical and observational groups.
Main Results:
- Children who underwent surgery for CLM showed significantly reduced lung function (FEV1, FEV1/FVC, FEF25%-75%) compared to controls.
- Children managed by observation also exhibited reduced FEV1/FVC and FEF25%-75%, though FEV1 and FVC remained normal.
- Exercise tolerance was significantly impaired in both groups, with a notable percentage scoring below -1 SDS in both surgical and observational cohorts. Physical growth was normal in all participants.
Conclusions:
- Children with CLM, especially those who have undergone surgery, are at risk for diminished lung function and exercise capacity.
- The study suggests that a watchful waiting approach may be appropriate for children with asymptomatic CLM, given the limited pulmonary morbidity observed in this subgroup.
- Long-term monitoring is essential for children with CLM to manage potential respiratory and exercise limitations.
Objective:
To improve counseling on congenital lung malformations (CLM) by describing long-term outcomes of children either operated on or managed by observation.
Study Design:
We analyzed lung function (spirometry), exercise tolerance (Bruce treadmill), and physical growth of 8-year-old children with CLM who participated in our longitudinal prospective follow-up program. The data are shown as median standard deviation scores (SDS) with IQR, or estimated marginal means (95% CI) on the basis of general linear models.
Results:
Twenty-nine (48%) of the 61 children had required surgery at a median age of 108 (IQR: 8-828) days, and 32 (52%) were managed by observation. In the surgery group, all lung function measurements (except for forced vital capacity [FVC]) were significantly below 0 SDS, with median FEV1 -1.07 (IQR: -1.70 to -0.56), FEV1 /FVC -1.49 (-2.62 to -0.33), and FEF25%-75% -1.95 (-2.57 to -0.63) (all P < 0.001). Children in the observation group had normal FEV1 and FVC, whereas FEV1 /FVC (-0.81 (-1.65 to -0.14)) and FEF25%-75% (-1.14 (-1.71 to -0.22)) were significantly below 0 SDS (both P < 0.001). Mean exercise tolerance was significantly below 0 SDS in both groups (observation: -0.85 (95% CI: -1.30 to -0.41); surgery: -1.25 (-1.69 to -0.80)); eight (28%) children in the observation group and ten (40%) in the surgery group scored <-1 SDS. Physical growth was normal in both groups.
Conclusion:
Children with CLM may be at risk for reduced lung function and exercise tolerance, especially those who required surgery. As little pulmonary morbidity was found in children with asymptomatic CLM, this study supports a watchful waiting approach in this group.
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