Abstract
BackgroundAngiolymphoid 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.ReportWe 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.ConclusionALHE 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.
Highlights
Angiolymphoid hyperplasia with eosinophilia (ALHE) is characterized clinically by single to multiple red brown dome shaped papules or subcutaneous nodules located mainly in the head and neck [1,2,3,4]
The lesions are characterized by a reactive proliferation of small blood vessels, often surrounding a muscular artery, with peripheral inflammatory infiltrates consisting of mononuclear cells and eosinophils
The purpose of our study is to report the first documented case of ALHE developing after the diagnosis of peripheral T-cell lymphoma with T-cell receptor gene rearrangements showing monoclonality in both the lymphoma and the vascular lesion
Summary
ALHE is characterized clinically by single to multiple red brown dome shaped papules or subcutaneous nodules located mainly in the head and neck [1,2,3,4]. The lesions are characterized by a reactive proliferation of small blood vessels, often surrounding a muscular artery, with peripheral inflammatory infiltrates consisting of mononuclear cells and eosinophils. 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. It is unclear whether it is a reactive or neoplastic process. 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. 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
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