ArticleCancer medicine2020
Integrative clinical and biopathology analyses to understand the clinical heterogeneity of infantile rhabdomyosarcoma: A report from the French MMT committee.
Article in Cancer medicine, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed, 33 citations in OpenAlex.
- Paediatric Therapeutic Development Workshop on rhabdomyosarcoma.British journal of cancer · 2026Review
- MYOD1 mutation drives cancer stem cell pathways and therapy-resistance in spindle cell/sclerosing rhabdomyosarcoma.Nature communications · 2026Article
- Review
- Current management of neurotrophic receptor tyrosine kinase fusion-positive sarcoma: an updated review.Japanese journal of clinical oncology · 2025Review
- Single cell transcriptomic profiling identifies tumor-acquired and therapy-resistant cell states in pediatric rhabdomyosarcoma.Nature communications · 2024Article
- Biological and therapeutic insights from animal modeling of fusion-driven pediatric soft tissue sarcomas.Disease models & mechanisms · 2024Review
- FGFR1 fusions as a novel molecular driver in rhabdomyosarcoma.Genes, chromosomes & cancer · 2024Article
- Review
- [Special mesenchymal neoplasms of the head and neck: Update from the 2022 WHO].Pathologie (Heidelberg, Germany) · 2023Review
- Clinical Characteristics, Treatment Considerations, and Outcomes of Infants with Rhabdomyosarcoma.Cancers · 2023Review
- VGLL2-NCOA2 leverages developmental programs for pediatric sarcomagenesis.Cell reports · 2023Article
- Recurrent VGLL3 fusions define a distinctive subset of spindle cell rhabdomyosarcoma with an indolent clinical course and striking predilection for the head and neck.Genes, chromosomes & cancer · 2022Article
- ZFP64::NCOA3 gene fusion defines a novel subset of spindle cell rhabdomyosarcoma.Genes, chromosomes & cancer · 2022Article
- The Molecular Biology of Soft Tissue Sarcomas: Current Knowledge and Future Perspectives.Cancers · 2022Review
- Spindle cell/sclerosing rhabdomyosarcoma with DCTN1::ALK fusion: broadening the molecular spectrum with potential therapeutic implications.Virchows Archiv : an international journal of pathology · 2022Article
- Establishment and Characterization of a Cell Line (S-RMS1) Derived from an Infantile Spindle Cell Rhabdomyosarcoma withInternational journal of molecular sciences · 2021Article
- The Evolving Role of Radiotherapy for Pediatric Cancers With Advancements in Molecular Tumor Characterization and Targeted Therapies.Frontiers in oncology · 2021Review
- Integrative clinical and biopathology analyses to understand the clinical heterogeneity of infantile rhabdomyosarcoma: A report from the French MMT committee.Cancer medicine · 2020Article
- Recent advances in understanding and managing pediatric rhabdomyosarcoma.F1000Research · 2020Review
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Authors and funding
19 authors at 14 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundRhabdomyosarcoma (RMS) in infants is a particular entity with various clinical presentations and outcomes. To better understand the clinical heterogeneity of RMS in infants, an integrative clinical, histological, and molecular analysis was performed.
methodsFrom 1989 to 2015, 37 infants aged less than 6 months with a diagnosis of RMS and archival tumor materials were identified in France. Clinical data, central pathologic review, and molecular profile including RNA sequencing were analyzed.
resultsNineteen patients (51%) had embryonal RMS (ERMS) (including three highly differentiated ERMS with PTCH deletion), eight (22%) had spindle cell RMS (SRMS) (three VGLL2-, one NTRK-, and two (B)RAF-fusions), six (16%) had alveolar RMS (ARMS) (all FOXO1- or PAX3-fusion), two had unclassified RMS, and two poorly differentiated RMS were retrospectively diagnosed as rhabdoid tumors (RT) with loss of INI1 expression. The two RT patients died of rapid disease progression. Five-year event-free (EFS) and overall survival (OS) for RMS were 62% (95%CI, 47-82) and 52% (95%CI, 37-72). Eleven patients (31%) relapsed and four (11%) had primary refractory disease (all ERMS). In univariate analysis, EFS and OS were only associated with histology subtype, with 100% survival of known fusion-positive SRMS. RNA cluster expression showed three main clusters: ARMS, ERMS, and "VGLL2-fusion" cluster, consisting of SRMS and ERMS.
conclusionsBiopathology findings from this study support the different prognosis of infantile RMS. New fusion-positive SRMS has a very good outcome which may allow more conservative treatment in the future.
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