Literature DB >> 34089379

The role of molecular diagnostics in aneurysmal and simple bone cysts - a prospective analysis of 19 lesions.

Jože Pižem1, Daja Šekoranja2, Alenka Matjašič2, Andrej Zupan2, Emanuela Boštjančič2, Katarina A Limpel Novak3, Vladka Salapura3,4, Blaž Mavčič4,5, Barbara Gazič6, Katarina Dimnik2.   

Abstract

Aneurysmal (ABC) and simple bone cysts (SBC) have been traditionally distinguished by radiological and histopathological features. However, there is some radiological and histopathological overlap between ABC and SBC. ABC is characterised by USP6 fusions while, recently, NFATC2 fusions have been found in a large proportion of SBC. Identifying these fusions may assist in confirming the diagnosis of either ABC or SBC. To elaborate the potential benefit of molecular testing, we report a prospective series of 19 consecutive bone cysts with comprehensive radiological, histopathological and molecular diagnostics. Integrating radiological, histopathological and molecular findings, 11 cysts were diagnosed as SBC and 8 as ABC. Radiologically, 6 of 11 SBC and 6 of 8 ABC were diagnosed as ABC. Fibrin-like collagen deposits were identified in 8 of 11 (73%) SBC and 3 of 8 (38%) ABC. Nodular fasciitis-like areas were identified in 6 of 8 (75%) ABC and in 7 of 11 (64%) SBC. A USP6 fusion was identified in all 8 ABC, including a novel RBM5-USP6 fusion. An NFATC2 fusion was found in 7 of 11 SBC (FUS-NFATC2 fusion in 5 and EWSR1-NFATC2 in 2 cases). There is radiological and histopathological overlap between SBC and ABC in a significant proportion of cases. A diagnosis of ABC is frequently suggested radiologically in SBC, and fibrin-like deposits, thought to be specific for SBC, may be found in some ABC. Molecular testing may significantly improve diagnostic accuracy in bone cysts.

Entities:  

Keywords:  Aneurysmal bone cyst; NFATC2; RBM5; RNA sequencing; Simple bone cyst; USP6

Year:  2021        PMID: 34089379     DOI: 10.1007/s00428-021-03130-5

Source DB:  PubMed          Journal:  Virchows Arch        ISSN: 0945-6317            Impact factor:   4.064


  11 in total

1.  The nature of the characteristic cementum-like matrix deposits in the walls of simple bone cysts.

Authors:  Daniel Baumhoer; Jan Smida; Michaela Nathrath; Gernot Jundt
Journal:  Histopathology       Date:  2011-09       Impact factor: 5.087

2.  Cementum-like matrix in solitary bone cysts: a unique and characteristic but yet underrecognized feature of promising diagnostic utility.

Authors:  Muhammad Usman Tariq; Nasir Ud Din; Zubair Ahmad; Naila Kayani; Rashida Ahmed
Journal:  Ann Diagn Pathol       Date:  2013-10-01       Impact factor: 2.090

3.  MR imaging of simple bone cysts in children: not so simple.

Authors:  R Margau; P Babyn; W Cole; C Smith; F Lee
Journal:  Pediatr Radiol       Date:  2000-08

4.  Prevalence of somatic and germline mutations of Fumarate hydratase in uterine leiomyomas from young patients.

Authors:  Chengbao Liu; Jessica Dillon; Anna L Beavis; Yuehua Liu; Kara Lombardo; Amanda N Fader; Chien-Fu Hung; Tzyy-Choou Wu; Russell Vang; Jairo E Garcia; Deyin Xing
Journal:  Histopathology       Date:  2019-09-28       Impact factor: 5.087

5.  Novel ASAP1-USP6, FAT1-USP6, SAR1A-USP6, and TNC-USP6 fusions in primary aneurysmal bone cyst.

Authors:  Daja Šekoranja; Andrej Zupan; Blaž Mavčič; David Martinčič; Vladka Salapura; Žiga Snoj; Ana K Limpel Novak; Jože Pižem
Journal:  Genes Chromosomes Cancer       Date:  2020-02-08       Impact factor: 5.006

6.  USP6 and CDH11 oncogenes identify the neoplastic cell in primary aneurysmal bone cysts and are absent in so-called secondary aneurysmal bone cysts.

Authors:  Andre M Oliveira; Antonio R Perez-Atayde; Carrie Y Inwards; Fabiola Medeiros; Victoria Derr; Bae-Li Hsi; Mark C Gebhardt; Andrew E Rosenberg; Jonathan A Fletcher
Journal:  Am J Pathol       Date:  2004-11       Impact factor: 4.307

7.  Anchored multiplex PCR for targeted next-generation sequencing reveals recurrent and novel USP6 fusions and upregulation of USP6 expression in aneurysmal bone cyst.

Authors:  Natalya V Guseva; Omar Jaber; Munir R Tanas; Aaron A Stence; Ramakrishna Sompallae; Jenna Schade; Allison N Fillman; Benjamin J Miller; Aaron D Bossler; Deqin Ma
Journal:  Genes Chromosomes Cancer       Date:  2016-12-02       Impact factor: 5.006

Review 8.  WHO classification of soft tissue tumours: an update based on the 2013 (4th) edition.

Authors:  Vickie Y Jo; Christopher D M Fletcher
Journal:  Pathology       Date:  2014-02       Impact factor: 5.306

9.  FUS-NFATC2 or EWSR1-NFATC2 Fusions Are Present in a Large Proportion of Simple Bone Cysts.

Authors:  Jože Pižem; Daja Šekoranja; Andrej Zupan; Emanuela Boštjančič; Alenka Matjašič; Blaž Mavčič; Juan A Contreras; Barbara Gazič; David Martinčič; Žiga Snoj; Katarina A Limpel Novak; Vladka Salapura
Journal:  Am J Surg Pathol       Date:  2020-12       Impact factor: 6.394

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