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Spindle Epithelial Tumor With Thymus-Like Differentiation in an Infant.
Kei Yu Chiu1, Phillip Sale2,3, Adrian Charles4
1Department of Paediatric and Adolescent Oncology/Haematology, Perth Children's Hospital, Nedlands, Western Australia, Australia.
Pediatrics
|July 14, 2026
Summary
Spindle epithelial tumor with thymus-like differentiation (SETTLE) is a rare cancer. This report details a unique infant case of sublingual SETTLE, highlighting diagnostic challenges and its unusual location.
Area of Science:
- Oncology
- Pathology
- Pediatric Neoplasms
Background:
- Spindle epithelial tumor with thymus-like differentiation (SETTLE) is an exceptionally rare malignant neoplasm.
- SETTLE predominantly affects the thyroid gland, with limited literature on extrathyroidal presentations.
- Extrathyroidal SETTLE cases, particularly in infants, are virtually undocumented.
Purpose of the Study:
- To report a novel case of sublingual SETTLE in an infant.
- To discuss the diagnostic complexities associated with extrathyroidal SETTLE.
- To contribute to the understanding of rare pediatric neoplasms and their clinical behavior.
Main Methods:
- Case report of an infant presenting with a sublingual mass.
- Histopathological examination of incisional biopsy and subsequent gross total resection.
- Specialist pathology review for definitive diagnosis of SETTLE.
Main Results:
- Initial histopathology suggested chronic sialadenitis, complicating the diagnosis.
- Gross total resection revealed a low-grade spindle epithelial cell neoplasm.
- Specialist review confirmed the diagnosis as SETTLE, noting the patient's young age and extrathyroidal tumor location.
Conclusions:
- This case represents the youngest patient diagnosed with SETTLE and the first reported instance of sublingual SETTLE.
- Accurate histopathological evaluation and specialist consultation are crucial for diagnosing rare neoplasms like SETTLE.
- The findings expand the known spectrum of SETTLE presentation and location, emphasizing the need for vigilance in pediatric oncology.
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