ArticleBMC pulmonary medicine2025
Lymphatic malformations involving the thorax in children: a retrospective cohort study.
Article in BMC pulmonary medicine, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.
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Who cites it
3 citing papers in PubMed.
- Kaposiform lymphangiomatosis-the effects of long-term treatment with sirolimus: case series study and review of the literature.Frontiers in medicine · 2026Article
- Long-term low-dose sirolimus therapy and successful discontinuation in an adult with kaposiform lymphangiomatosis and disseminated intravascular coagulation: a case report.Frontiers in medicine · 2026Article
- Genotype-directed Targeted Therapy for Pediatric Vascular Malformations: A Systematic Review.Journal of Indian Association of Pediatric SurgeonsReview
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Authors and funding
8 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundLymphatic malformations (LMs) involving the thorax are rare, with limited clinical understanding. We aimed to summarize the classification, clinical features, treatment, and prognosis of thoracic LMs, and to improve disease management and patient outcomes.
methodsClinical data and follow-up data obtained from 42 patients with thoracic LMs were reviewed retrospectively at a single center in China.
resultsPatients were classified into 7 types: 1 with macrocystic LM, 3 with infancy primary chylothorax, 4 with primary lymphedema (PL), and 34 with complicated lymphatic anomalies (CLAs), including 18 with generalized lymphatic anomaly (GLA), 8 with kaposiform lymphangiomatosis (KLA), 6 with central conducting lymphatic anomaly (CCLA), and 2 with Gorham-Stout disease. The specific clinical manifestations included chylothorax (50%), white foamy/jelly-like sputum (47.6%), and plastic bronchitis (7.1%). Imaging findings revealed interlobular septal thickening in 20 patients (47.6%) and ground-glass opacity in 13 (31.0%). Improvements were observed in 16 patients with CLAs who were administered sirolimus, 2 with GLA who were administered sirolimus and bevacizumab, 1 with KLA who was administered trametinib, 6 with CCLA who underwent surgery, 3 with infancy primary chylothorax following dietary treatment, and 1 with macrocystic LM following sclerotherapy. Stabilization occurred in 7 patients (3 with CLAs and 4 with PL) postsurgery. Progression or death was observed in 4 patients with GLA and 3 patients with KLA.
conclusionsDifferent types of thoracic LMs have similar clinical features and imaging manifestations but vary in terms of treatment and prognosis.
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