Angiolymphoid hyperplasia with eosinophilia developing in a patient with history of peripheral T-cell lymphoma: evidence for multicentric T-cell lymphoproliferative process

Luis F Gonzalez-Cuyar¹ , Fabio Tavora¹ , X Frank Zhao¹ , Guanghua Wang³ , Aaron Auerbach² , Nadine Aguilera² and Allen P Burke¹

¹Department of Pathology, University of Maryland School of Medicine, Baltimore, Maryland, USA

²Department of Hematopathology, Armed Forces Institute of Pathology, Washington DC, USA

³Department of Molecular Diagnostics, Armed Forces Institute of Pathology, Washington DC, USA

author email corresponding author email

Diagnostic Pathology 2008, 3:22doi:10.1186/1746-1596-3-22


Published:	29 May 2008

Abstract

Background

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a vasocentric process characterized by infiltrates of lymphocytes and eosinophils, usually affecting the muscular arteries of the head and neck. Currently it is unclear whether it is a reactive or neoplastic process.

Report

We present a 61-year-old African American male with a twenty year history of superficial skin patches involving the head and neck region. An excisional biopsy of a right submental lymph node revealed an atypical T-cell lymphocytic process, diagnosed as peripheral T-cell lymphoma after immunophenotyping and molecular studies. Three months later the patient underwent a biopsy of a left temporal nodule that was diagnosed as ALHE. Subsequently, at two year follow-up, the patient was diagnosed with Mycosis Fungoides. Polymerase chain reaction for T cell receptor gamma showed the same T-cell receptor gene rearrangement in both the temporal mass and the right submental lymph node.

Conclusion

ALHE with molecular evidence of monoclonality is extremely unusual, as is the association with nodal peripheral T-cell nodal lymphoma. The findings of this case support our hypothesis that ALHE might be an early form of T-cell lymphoma.