ArticleJournal of children's orthopaedics2020
The correlation between spinal and chest wall deformities and pulmonary function in Marfan syndrome.
Article in Journal of children's orthopaedics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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Who cites it
6 citing papers in PubMed, 14 citations in OpenAlex.
- Article
- Unique Features of Cardiovascular Involvement and Progression in Children with Marfan Syndrome Justify Dedicated Multidisciplinary Care.Journal of cardiovascular development and disease · 2024Review
- Integrated DNA methylation analysis reveals a potential role for PTPRN2 in Marfan syndrome scoliosis.JOR spine · 2024Article
- Effects of spinal deformities on lung development in children: a review.Journal of orthopaedic surgery and research · 2023Review
- Swallowing and quality of life in individuals with Marfan syndrome: a cross-sectional study.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2022Article
- Is physical activity a future therapy for patients with Marfan syndrome?Orphanet journal of rare diseases · 2022Review
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Authors and funding
4 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
purposeScoliosis, chest wall deformities and pulmonary involvement are common features of Marfan syndrome (MFS). We aimed to assess the impact of spinal and chest wall deformities on pulmonary function in paediatric MFS patients with a surgically managed spinal deformity.
methodsIn this multicentre retrospective study, spirometry, lung volumes and radiographic imaging were performed on 26 MFS patients between the ages of seven and 18 years who were undergoing planned spinal fusion surgery for spinal deformity. A correlation analysis assessed the relationship between radiographic measurements of spinal and chest wall deformities and predicted total lung capacity (TLC), forced vital capacity (FVC) and the ratio between forced expiratory volume in one second and FVC (FEV1/FVC).
resultsIn total, 18 patients (70%) had impaired pulmonary function. Thoracic kyphosis (mean 19.3°; -32° to 54°) had a strong positive correlation with FEV1/FVC (r = 0.65; p < 0.001). Significant decrease in FEV1/FVC below 80% occurred at kyphosis under 15° (p = 0.004). Kyphosis had a moderate negative correlation with FVC (r = -0.43; p = 0.03). Chest wall deformity had a strong negative correlation with FEV1/FVC (r = -0.61; p = 0.001). The magnitude of the thoracic curve (mean 55.2°; 28° to 92°) had a significant moderate negative correlation with TLC (r = -0.45; p = 0.04).
conclusionIn MFS, three factors correlate with decreased pulmonary function measures: hypokyphosis, increasing chest wall deformity and increasing coronal curve magnitude. Hypokyphosis and increased chest wall deformity correlated with diminished FEV1/FVC; increasing thoracic spinal curvature with diminished TLC. Further analysis with a larger cohort will help better define the relationship between these deformities and pulmonary function in this unique population. LEVEL OF EVIDENCE: IV.
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