Prevalence of locoregional and distant lymph node metastases in children and adolescents/young adults with soft tissue sarcomas: a Bayesian meta-analysis of proportions
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Elsevier
Abstract
Background: The presence of both regional and distant lymph node metastases (LNM) in paediatric and adolescent/
young adult (AYA) patients with soft tissue sarcomas (STS) significantly impacts clinical outcomes. However, reported
rates of LNM vary widely across the literature and are often accompanied by substantial uncertainty. We
aimed to quantitatively synthesise global proportions of LNM across different histological subtypes and tumour sites
in this population.
Methods: In this meta-analysis, we systematically searched MEDLINE, Scopus, and Web of Science from inception
until May 1, 2024 (updated on June 1, 2025) for studies published in English that reported LNM rates in patients
with STS aged 0–21 years. Eligible study designs included cohort studies, case-control studies, case series, and
randomised controlled trials. Patient-level data were not requested from study authors. LNM had to be confirmed
clinically, by imaging, or histologically. We excluded reviews, editorials, and case reports with fewer than three
patients. We conducted a random-effects Bayesian meta-analysis using a logit transformation to synthesise LNM
proportions. The posterior distributions of LNM prevalence were summarised by the posterior mean and 95%
credible intervals (CrIs). Study quality was assessed across seven domains: confounding, participant selection,
exposure classification, deviations from intended exposures, missing data, outcome measurement, and selective
reporting.
Findings: Of 3969 records screened, 263 articles were included in the data synthesis. These comprised 147 studies on
rhabdomyosarcoma (RMS), 106 on non-rhabdomyosarcoma soft tissue sarcoma (NRSTS), and 10 on mixed RMS/
NRSTS cohorts, representing 53,093 patients with STS. The pooled posterior mean proportion of LNM in patients
with RMS (n = 41,547) was 0.228 (95% CrI: 0.202–0.255), with the highest rates observed in patients with alveolar
RMS (posterior mean proportion: 0.370; 95% CrI: 0.276–0.473). Subgroup analysis by RMS primary site revealed the
highest LNM rates in the perianal/perineal region (0.466; 95% CrI: 0.397–0.537), extremity (0.281; 0.210–0.363), and
non-parameningeal head/neck region (0.259; 0.167–0.376). Among patients with NRSTS (n = 11,546), the pooled
posterior mean proportion of LNM was 0.111 (95% CrI: 0.092–0.133), with desmoplastic small round cell tumour
(0.440; 0.335–0.552), clear cell sarcoma (0.212; 0.163–0.275), and malignant rhabdoid tumour (0.199; 0.141–0.273)
showing the highest rates. Most analyses had moderate-to-high heterogeneity (95% CrI for tau: 0.7443–1.1139).
Interpretation: Our Bayesian meta-analysis synthesises global evidence on the prevalence of LNM in paediatric and
AYA patients with STS, highlighting the significant heterogeneity in LNM rates by histological subtype, particularly
in NRSTS, and by tumour location, especially in RMS. Future studies should aim to standardise lymph node staging
protocols and reporting practices to improve classification accuracy and enhance comparability across studies.
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