Abstract
Introduction
Evidence on exercise training adaptations in adults with a history of congenital pulmonary airway malformation (CPAM) treated with early-life lobectomy is scarce. This case report describes the clinical and physiological responses to the addition of aerobic training to an ongoing resistance training program in an individual with markedly reduced forced vital capacity (FVC) and forced expiratory volume in the first second (FEV1) and elevated FEV1/FVC, suggestive of a restrictive ventilatory impairment, contributing novel clinical data in a rare population.Main symptoms and clinical findings
A male patient with a history of CPAM, treated with right upper lobectomy at 6 months of age, presented in adulthood with a restrictive ventilatory impairment. Additional findings included minor arrhythmia, pectus excavatum, and mild mitral valve prolapse.Diagnosis, intervention, and outcomes
The main diagnosis was prior CPAM status post lobectomy with persistent ventilatory limitation. The patient underwent a structured resistance training program with the addition of once-weekly aerobic training. After the intervention, reductions in submaximal heart rate during standardized walking were observed (-7.1% at 1 km·h-1, 0% grade and -6.5% at 1 km·h-1, 8.7% grade), suggesting potencial improved cardiovascular efficiency during standardized submaximal exercise. There were also changes in neuromuscular performance (estimated 1RM: bench press 40.8 to 43.2 kg; Δ +2.4 kg, +5.9%; leg press 112.5 to 120.0 kg; Δ +7.5 kg, +6.7%) and respiratory-related quality of life, as assessed by the St. George's Respiratory Questionnaire (SGRQ, total score 23.42 to 18.98; Δ -4.4 points; symptoms 19.97 to 8.56; Δ -11.4 points; impacts 17.40 to 12.08; Δ -5.3 points), while the activity domain remained essentially unchanged (35.41 to 35.47; Δ +0.1 point), indicating clinically relevant difference for total score domain in similar clinical populations, but causality cannot be inferred.Conclusion
Under supervised conditions, the addition of once-weekly aerobic training to ongoing resistance training was feasible and generally well tolerated in this patient, and was associated with favorable changes in submaximal heart rate response, estimated strength, and selected SGRQ domains. These observations are hypothesis-generating only.