Literature DB >> 32506523

Soft tissue tumors characterized by a wide spectrum of kinase fusions share a lipofibromatosis-like neural tumor pattern.

Yu-Chien Kao1,2, Albert J H Suurmeijer3, Pedram Argani4, Brendan C Dickson5, Lei Zhang6, Yun-Shao Sung6, Narasimhan P Agaram6, Christopher D M Fletcher7, Cristina R Antonescu6.   

Abstract

Gene fusions resulting in oncogenic activation of various receptor tyrosine kinases, including NTRK1-3, ALK, and RET, have been increasingly recognized in soft tissue tumors (STTs), displaying a wide morphologic spectrum and therefore diagnostically challenging. A subset of STT with NTRK1 rearrangements were recently defined as lipofibromatosis-like neural tumors (LPFNTs), being characterized by mildly atypical spindle cells with a highly infiltrative growth in the subcutis and expression of S100 and CD34 immunostains. Other emerging morphologic phenotypes associated with kinase fusions include infantile/adult fibrosarcoma and malignant peripheral nerve sheath tumor-like patterns. In this study, a large cohort of 73 STT positive for various kinase fusions, including 44 previously published cases, was investigated for the presence of an LPFNT phenotype, to better define the incidence of this distinctive morphologic pattern and its relationship with various gene fusions. Surprisingly, half (36/73) of STT with kinase fusions showed at least a focal LPFNT component defined as >10%. Most of the tumors occurred in the subcutaneous tissues of the extremities (n = 25) and trunk (n = 9) of children or young adults (<30 years old) of both genders. Two-thirds (24/36) of these cases showed hybrid morphologies with alternating LPFNT and solid areas of monomorphic spindle to ovoid tumor cells with fascicular or haphazard arrangement, while one-third (12/36) had pure LPFNT morphology. Other common histologic findings included lymphocytic infiltrates, staghorn-like vessels, and perivascular or stromal hyalinization, especially in hybrid cases. Mitotic activity was generally low (<4/10 high power fields in 81% cases), being increased only in a minority of cases. Immunoreactivity for CD34 (92% in hybrid cases, 89% in pure cases) and S100 (89% in hybrid cases, 64% in pure cases) were commonly present. The gene rearrangements most commonly involved NTRK1 (75%), followed by RET (8%) and less commonly NTRK2, NTRK3, ROS1, ALK, and MET.
© 2020 Wiley Periodicals LLC.

Entities:  

Keywords:  MET; NTRK; RET; lipofibromatosis-like neural tumor

Year:  2020        PMID: 32506523     DOI: 10.1002/gcc.22877

Source DB:  PubMed          Journal:  Genes Chromosomes Cancer        ISSN: 1045-2257            Impact factor:   5.006


  11 in total

Review 1.  Diagnosis of soft tissue tumors using immunohistochemistry as a surrogate for recurrent fusion oncoproteins.

Authors:  Margaret A Black; Gregory W Charville
Journal:  Semin Diagn Pathol       Date:  2021-11-03       Impact factor: 3.464

Review 2.  WHO Pathology: Highlights of the 2020 Sarcoma Update.

Authors:  Inga-Marie Schaefer; Alessandro Gronchi
Journal:  Surg Oncol Clin N Am       Date:  2022-05-31       Impact factor: 2.402

3.  Adult NTRK-rearranged spindle cell neoplasms of the viscera: with an emphasis on rare locations and heterologous elements.

Authors:  Jen-Wei Tsai; Jen-Chieh Lee; Tsung-Han Hsieh; Shih-Chiang Huang; Pei-Hang Lee; Ting-Ting Liu; Yu-Chien Kao; Ching-Di Chang; Te-Fu Weng; Chien-Feng Li; Jung-Chia Lin; Cher-Wei Liang; Yu-Li Su; Ian Yi-Feng Chang; Yu-Ting Wang; Nien-Yi Chang; Shih-Chen Yu; Jui-Chu Wang; Hsuan-Ying Huang
Journal:  Mod Pathol       Date:  2022-02-11       Impact factor: 8.209

4.  NTRK Fusions in a Sarcomas Series: Pathology, Molecular and Clinical Aspects.

Authors:  Vasiliki Siozopoulou; Elly Marcq; Koen De Winne; Koen Norga; Gertjan Schmitz; Valerie Duwel; Philippe Delvenne; Evelien Smits; Patrick Pauwels
Journal:  Pathol Oncol Res       Date:  2022-05-11       Impact factor: 2.874

5.  Superficial ALK-rearranged myxoid spindle cell neoplasm: a cutaneous soft tissue tumor with distinctive morphology and immunophenotypic profile.

Authors:  Steven D Billings; Jennifer S Ko; Josephine K Dermawan; Elizabeth M Azzato; John R Goldblum; Brian P Rubin
Journal:  Mod Pathol       Date:  2021-06-04       Impact factor: 7.842

6.  Unclassified low grade spindle cell sarcoma with storiform pattern characterized by recurrent novel EWSR1/FUS-NACC1 fusions.

Authors:  Cristina R Antonescu; Brendan C Dickson; Lei Zhang; Yun-Shao Sung; Christopher D Fletcher
Journal:  Mod Pathol       Date:  2021-04-15       Impact factor: 7.842

7.  Pediatric fibromyxoid soft tissue tumor with PLAG1 fusion: A novel entity?

Authors:  Catherine T Chung; Cristina R Antonescu; Brendan C Dickson; Rose Chami; Paula Marrano; Rong Fan; Mary Shago; Meera Hameed; Paul S Thorner
Journal:  Genes Chromosomes Cancer       Date:  2020-12-30       Impact factor: 5.006

8.  Novel BRAF gene fusions and activating point mutations in spindle cell sarcomas with histologic overlap with infantile fibrosarcoma.

Authors:  Alyssa J Penning; Alyaa Al-Ibraheemi; Michael Michal; Brandon T Larsen; Soo-Jin Cho; Christina M Lockwood; Vera A Paulson; Yajuan J Liu; Lukáš Plank; Karen Fritchie; Carol Beadling; Tanaya L Neff; Christopher L Corless; Erin R Rudzinski; Jessica L Davis
Journal:  Mod Pathol       Date:  2021-04-13       Impact factor: 8.209

9.  Low-grade non-intestinal-type sinonasal adenocarcinoma: a histologically distinctive but molecularly heterogeneous entity.

Authors:  Lisa M Rooper; Lester D R Thompson; Jeffrey Gagan; Jacqueline Siok Gek Hwang; Nyall R London; Michael W Mikula; Todd M Stevens; Justin A Bishop
Journal:  Mod Pathol       Date:  2022-03-23       Impact factor: 8.209

Review 10.  NTRK Fusions in Sarcomas: Diagnostic Challenges and Clinical Aspects.

Authors:  Vasiliki Siozopoulou; Evelien Smits; Koen De Winne; Elly Marcq; Patrick Pauwels
Journal:  Diagnostics (Basel)       Date:  2021-03-09
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