Complete response of metastatic alveolar soft part sarcoma in an adolescent female treated by combined immune checkpoint inhibitors
Youngeun Jung
Department of Pediatrics, Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Daegu, Republic of Korea
Search for more papers by this authorCorresponding Author
Ye Jee Shim
Department of Pediatrics, Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Daegu, Republic of Korea
Correspondence
Ye Jee Shim, Department of Pediatrics, Keimyung University School of Medicine, 1095 Dalgubeol-daero, Dalseo-gu, Daegu 42601, Republic of Korea.
Email: [email protected]
Search for more papers by this authorHye Ra Jung
Department of Pathology, Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Daegu, Republic of Korea
Search for more papers by this authorHeung Sik Kim
Department of Pediatrics, Keimyung University School of Medicine, Keimyung University Daegu Dongsan Hospital, Daegu, Republic of Korea
Search for more papers by this authorYoungeun Jung
Department of Pediatrics, Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Daegu, Republic of Korea
Search for more papers by this authorCorresponding Author
Ye Jee Shim
Department of Pediatrics, Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Daegu, Republic of Korea
Correspondence
Ye Jee Shim, Department of Pediatrics, Keimyung University School of Medicine, 1095 Dalgubeol-daero, Dalseo-gu, Daegu 42601, Republic of Korea.
Email: [email protected]
Search for more papers by this authorHye Ra Jung
Department of Pathology, Keimyung University School of Medicine, Keimyung University Dongsan Hospital, Daegu, Republic of Korea
Search for more papers by this authorHeung Sik Kim
Department of Pediatrics, Keimyung University School of Medicine, Keimyung University Daegu Dongsan Hospital, Daegu, Republic of Korea
Search for more papers by this authorFunding information:
Bisa Research Grant of Keimyung University (2019)
Supporting Information
| Filename | Description |
|---|---|
| pbc29787-sup-0001-FigureS1.docx439.7 KB | Supporting Figure S1 (A) The biopsied specimen shows solid sheets of large polygonal cells with abundant pinkish cytoplasm and round, slightly dull nuclei (H&E stain, ×400). (B) PAS staining shows intracytoplasmic granules (black arrows) (PAS stain, ×1000). (C and D) The tumor cells are positively stained by TEF3 (C, ×400) and myogenin (D, ×400) |
Please note: The publisher is not responsible for the content or functionality of any supporting information supplied by the authors. Any queries (other than missing content) should be directed to the corresponding author for the article.
REFERENCES
- 1Portera CA Jr, Ho V, Patel SR, et al. Alveolar soft part sarcoma: clinical course and patterns of metastasis in 70 patients treated at a single institution. Cancer. 2001; 91(3): 585-591.
- 2Casanova M, Ferrari A, Bisogno G, et al. Alveolar soft part sarcoma in children and adolescents: a report from the Soft-Tissue Sarcoma Italian Cooperative Group. Ann Oncol. 2000; 11(11): 1445-1449.
- 3Ogose A, Yazawa Y, Ueda T, et al. Alveolar soft part sarcoma in Japan: multi-institutional study of 57 patients from the Japanese Musculoskeletal Oncology Group. Oncology. 2003; 65(1): 7-13.
- 4Wang H, Jacobson A, Harmon DC, et al. Prognostic factors in alveolar soft part sarcoma: a SEER analysis. J Surg Oncol. 2016; 113(5): 581-586.
- 5Chang X, Li Y, Xue X, Zhou H, Hou L. The current management of alveolar soft part sarcomas. Medicine (Baltimore). 2021; 100(31):e26805.
- 6O'Sullivan Coyne G, Naqash AR, Sankaran H, Chen AP. Advances in the management of alveolar soft part sarcoma. Curr Probl Cancer. 2021; 45(4):100775.
- 7Saerens M, Brusselaers N, Rottey S, Decruyenaere A, Creytens D, Lapeire L. Immune checkpoint inhibitors in treatment of soft-tissue sarcoma: a systematic review and meta-analysis. Eur J Cancer. 2021; 152: 165-182.
- 8Wilky BA, Trucco MM, Subhawong TK, et al. Axitinib plus pembrolizumab in patients with advanced sarcomas including alveolar soft-part sarcoma: a single-centre, single-arm, phase 2 trial. Lancet Oncol. 2019; 20(6): 837-848.
- 9D'Angelo SP, Mahoney MR, Van Tine BA, et al. Nivolumab with or without ipilimumab treatment for metastatic sarcoma (Alliance A091401): two open-label, non-comparative, randomised, phase 2 trials. Lancet Oncol. 2018; 19(3): 416-426.
- 10Barsouk A, Rawla P, Hadjinicolaou AV, Aluru JS, Barsouk A. Targeted therapies and immunotherapies in the treatment of esophageal cancers. Med Sci (Basel). 2019; 7(10): 100.
- 11Reck M, Borghaei H, O'Byrne KJ. Nivolumab plus ipilimumab in non-small-cell lung cancer. Future Oncol. 2019; 15(19): 2287-2302.
- 12Conley AP, Trinh VA, Zobniw CM, et al. Positive tumor response to combined checkpoint inhibitors in a patient with refractory alveolar soft part sarcoma: a case report. J Glob Oncol. 2018; 4: 1-6. https://doi.org/10.1200/jgo.2017.009993
- 13Conry A, Peters M, Fried DB, et al. Complete response to dual immunotherapy in a young adult with metastatic alveolar soft part sarcoma enabled by a drug recovery program in a community practice. J Adolesc Young Adult Oncol. 2020; 9(3): 449-452.
