Document Detail


Epithelioid hemangioma of bone and soft tissue: a reappraisal of a controversial entity.
MedLine Citation:
PMID:  21948309     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
BACKGROUND: The controversy surrounding diagnosis of an epithelioid hemangioma (EH), particularly when arising in skeletal locations, stems not only from its overlapping features with other malignant vascular neoplasms, but also from its somewhat aggressive clinical characteristics, including multifocal presentation and occasional lymph node involvement. Specifically, the distinction from epithelioid hemangioendothelioma (EHE) has been controversial. The recurrent t(1;3)(p36;q25) chromosomal translocation, resulting in WWTR1-CAMTA1 fusion, recently identified in EHE of various anatomic sites, but not in EH or other epithelioid vascular neoplasms, suggests distinct pathogeneses.
QUESTION/PURPOSES: We investigated the clinicopathologic and radiologic characteristics of bone and soft tissue EHs in patients treated at our institution with available tissue for molecular testing.
PATIENTS AND METHODS: Seventeen patients were selected after confirming the pathologic diagnosis and fluorescence in situ hybridization analysis for the WWTR1 and/or CAMTA1 rearrangements. Four patients had multifocal presentation. Most patients with EH of bone were treated by intralesional curettage. None of the patients died of disease and only four patients had a local recurrence.
RESULTS: Our results, using molecular testing to support the pathologic diagnosis of EH, reinforce prior data that EH is a benign lesion characterized by an indolent clinical course with an occasional multifocal presentation and rare metastatic potential to locoregional lymph nodes.
CONCLUSION: These findings highlight the importance of distinguishing EH from other malignant epithelioid vascular tumors as a result of differences in their management and clinical outcome.
LEVEL OF EVIDENCE: Level IV, prognostic study. See Guidelines for Authors for a complete description of levels of evidence.
Authors:
Costantino Errani; Lei Zhang; David M Panicek; John H Healey; Cristina R Antonescu
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't     Date:  2011-09-24
Journal Detail:
Title:  Clinical orthopaedics and related research     Volume:  470     ISSN:  1528-1132     ISO Abbreviation:  Clin. Orthop. Relat. Res.     Publication Date:  2012 May 
Date Detail:
Created Date:  2012-03-29     Completed Date:  2012-05-15     Revised Date:  2013-11-06    
Medline Journal Info:
Nlm Unique ID:  0075674     Medline TA:  Clin Orthop Relat Res     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1498-506     Citation Subset:  AIM; IM    
Affiliation:
Department of Surgery, Memorial Sloan-Kettering Cancer Center, New York, NY, USA.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Adult
Aged
Aged, 80 and over
Bone Neoplasms / diagnosis*,  genetics
Calcium-Binding Proteins / genetics
Chromosomes, Human, Pair 1
Chromosomes, Human, Pair 3
DNA, Neoplasm / analysis
Diagnosis, Differential
Female
Gene Fusion
Hemangioendothelioma, Epithelioid / diagnosis*,  genetics
Humans
In Situ Hybridization, Fluorescence
Intracellular Signaling Peptides and Proteins / genetics
Male
Middle Aged
Soft Tissue Neoplasms / diagnosis*,  genetics
Trans-Activators / genetics
Translocation, Genetic
Vascular Neoplasms / diagnosis,  genetics
Young Adult
Grant Support
ID/Acronym/Agency:
P01 CA047179/CA/NCI NIH HHS; P01 CA047179-15A2/CA/NCI NIH HHS; P50 CA 140146-01/CA/NCI NIH HHS; P50 CA140146/CA/NCI NIH HHS
Chemical
Reg. No./Substance:
0/CAMTA1 protein, human; 0/Calcium-Binding Proteins; 0/DNA, Neoplasm; 0/Intracellular Signaling Peptides and Proteins; 0/Trans-Activators; 0/WWTR1 protein, human
Comments/Corrections

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