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Myositis ossificans-like soft tissue aneurysmal bone cyst: a clinical, radiological, and pathological study of seven cases with COL1A1-USP6 fusion and a novel ANGPTL2-USP6 fusion.

Authors :
Zhang L
Hwang S
Benayed R
Zhu GG
Mullaney KA
Rios KM
Sukhadia PY
Agaram N
Zhang Y
Bridge JA
Healey JH
Athanasian EA
Hameed M
Source :
Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc [Mod Pathol] 2020 Aug; Vol. 33 (8), pp. 1492-1504. Date of Electronic Publication: 2020 Mar 10.
Publication Year :
2020

Abstract

Herein we described the clinical, radiological, histological, and molecular characteristics of seven soft tissue aneurysmal bone cysts (STABCs) diagnosed and managed at a tertiary cancer center and to elucidate their relationship with myositis ossificans (MO). All cases had established imaging and histopathological diagnosis of STABC and were subject to fluorescence in situ hybridization (FISH) for USP6 rearrangement and Archer® FusionPlex® targeted RNA sequencing (RNASeq) analysis to identify the fusion partner. A thorough literature review of STABC and MO was conducted. The patients presented with painful masses unpreceded by trauma, occurring most commonly in the deep soft tissue of the thigh/gluteus (4/7), and also in the supraclavicular region, the axilla, and the hand. On imaging, the lesions were frequently associated with peripheral calcification on conventional radiographs and CT (6/7), cystic components on ultrasound, as well as perilesional edema (7/7) and fluid levels (3/7) on MRI. Bone scan (1/1) showed intense radiotracer uptake. Histologically, 6/7 cases demonstrated zonal arrangements reminiscent of MO. USP6 rearrangement was found in all seven cases by FISH and/or RNASeq. RNASeq further detected COL1A1-USP6 fusion in six cases and a novel ANGPTL2-USP6 fusion in one case. Four patients underwent resection of the tumors and were disease free at their last follow-up. Three patients who underwent incisional or needle biopsies had no evidence of disease progression on imaging studies. In conclusion, the clinical, radiological, and pathological overlap between STABC and MO suggests that they are closely related entities. A novel fusion ANGPTL2-USP6 is associated with distinct clinical and pathological presentation.

Details

Language :
English
ISSN :
1530-0285
Volume :
33
Issue :
8
Database :
MEDLINE
Journal :
Modern pathology : an official journal of the United States and Canadian Academy of Pathology, Inc
Publication Type :
Academic Journal
Accession number :
32157177
Full Text :
https://doi.org/10.1038/s41379-020-0513-4