Abstract
Spindle cell/sclerosing rhabdomyosarcoma (ssRMS) with FUS-TFCP2 translocation is a rare, aggressive RMS subtype often involving facial and pelvic bones and showing poor response to standard therapy. The FUS-TFCP2 fusion drives ALK gene activation and overexpression, suggesting ALK as a therapeutic target, though clinical use of ALK inhibitors remains limited in this context. We report a case of mandibular ssRMS with FUS-TFCP2 fusion treated with the third-generation ALK inhibitor Lorlatinib, resulting in a marked clinical response. We also review the potential utility of ALK-targeted therapies in managing FUS-TFCP2 fusion-positive ssRMS and support further exploration of ALK inhibition in this subset.
| Original language | English |
|---|---|
| Pages (from-to) | e29-e34 |
| Journal | Journal of Pediatric Hematology/Oncology |
| Volume | 48 |
| Issue number | 1 |
| DOIs | |
| State | Published - 1 Jan 2026 |
Keywords
- ALK inhibitor
- molecular alterations
- rhabdomyosarcoma
- spindle cell sclerosing
- target therapy
- Aminopyridines/therapeutic use
- Pyrazoles
- Humans
- Male
- Lactams/therapeutic use
- Molecular Targeted Therapy
- Repressor Proteins/genetics
- Rhabdomyosarcoma/genetics
- Lactams, Macrocyclic/therapeutic use
- Transcription Factors
- Oncogene Proteins, Fusion/genetics
- RNA-Binding Protein FUS/genetics
- DNA-Binding Proteins
- FUS-TFCP2
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