Molecular characterization of epithelioid haemangioendotheliomas identifies novel WWTR1-CAMTA1 fusion variants

Histopathology. 2015 Nov;67(5):699-708. doi: 10.1111/his.12697. Epub 2015 May 14.

Abstract

Aims: Epithelioid haemangioendothelioma (EHE) is a malignant vascular neoplasm. Subsets have been characterized previously by translocations resulting in either WWTR1-CAMTA1 or YAP1-TFE3 fusion. We sought to develop molecular and immunohistochemical (IHC) assays to aid in the diagnosis and characterization of EHE.

Methods and results: Fifty-two formalin-fixed, paraffin-embedded (FFPE) cases diagnosed between 2002 and 2014 were retrieved from the pathology files of our institutions. Reverse transcription-polymerase chain reaction (RT-PCR) assays were optimized to detect WWTR1-CAMTA1 and YAP1-TFE3 fusion transcripts in FFPE tissue and transcription factor E3 (TFE3) protein accumulation was examined by immunohistochemistry (IHC). RNA was extracted from 33 adequate samples, with more recent cases providing a greater yield of high quality RNA. Fourteen of 18 informative cases were positive for WWTR1-CAMTA1 fusion transcripts, four of which showed higher-grade cytological features termed by some as 'malignant EHE'. Novel in-frame fusion transcripts were identified in four cases by direct sequencing. IHC revealed variable nuclear TFE3 staining in six of 17 cases; three with patchy staining showed WWTR1-CAMTA1 fusion. One of 18 informative cases was positive for YAP1-TFE3 fusion and showed strong nuclear TFE3 staining by IHC.

Conclusions: This study confirms the high incidence of WWTR1-CAMTA1 and YAP1-TFE3 rearrangements in EHE and indicates that the staining pattern for TFE3 IHC is critical for specificity.

Keywords: CAMTA1; TFE3; WWTR1; YAP1; epithelioid; haemangioendothelioma; sarcoma; translocation.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptor Proteins, Signal Transducing / genetics
  • Adolescent
  • Adult
  • Aged
  • Basic Helix-Loop-Helix Leucine Zipper Transcription Factors / genetics
  • Calcium-Binding Proteins / genetics*
  • Female
  • Hemangioendothelioma, Epithelioid / genetics*
  • Humans
  • Immunohistochemistry
  • Intracellular Signaling Peptides and Proteins / genetics*
  • Male
  • Middle Aged
  • Oncogene Proteins, Fusion / genetics
  • Phosphoproteins / genetics
  • Reverse Transcriptase Polymerase Chain Reaction
  • Trans-Activators / genetics*
  • Transcription Factors
  • Transcriptional Coactivator with PDZ-Binding Motif Proteins
  • YAP-Signaling Proteins
  • Young Adult

Substances

  • Adaptor Proteins, Signal Transducing
  • Basic Helix-Loop-Helix Leucine Zipper Transcription Factors
  • CAMTA1 protein, human
  • Calcium-Binding Proteins
  • Intracellular Signaling Peptides and Proteins
  • Oncogene Proteins, Fusion
  • Phosphoproteins
  • TFE3 protein, human
  • Trans-Activators
  • Transcription Factors
  • Transcriptional Coactivator with PDZ-Binding Motif Proteins
  • WWTR1 protein, human
  • YAP-Signaling Proteins
  • YAP1 protein, human

Associated data

  • RefSeq/NM_0061064
  • RefSeq/NM_0065215
  • RefSeq/NM_0152153
  • RefSeq/NM_0154724