Angiolymphoid hyperplasia with eosinophilia affecting the scrotum: A rare case report with molecular evidence of T‐cell clonality

Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare, benign entity of unknown pathogenesis. It often presents as painful or pruritic intradermal or subcutaneous red to brown papules or nodules on the head and neck of young adults. A 38-year-old man had a gradually enlarging and mild pruritic plaque on the scrotum for half a year. Pathological findings showed dermal proliferation of anomalous blood vessels lined by plump endothelium with a significant perivascular inflammatory infiltrate composed of lymphocytes, histiocytes, scattered plasma cells and many eosinophils. They were consistent with the diagnosis of ALHE. In addition, the inflammatory infiltrate was analyzed by immunohistochemistry and T-cell receptor (TCR) gene rearrangement. They were mostly CD3(+) T cells and a monoclonal T-cell population. To the best of our knowledge, this is the first case of ALHE affecting the scrotum to be reported in the published work. We present this case to expand the anatomical distribution of this rare tumor. The molecular study of our case supports that ALHE might be a low-grade T-cell lymphoproliferative disorder.

Angiolymphoid hyperplasia with eosinophilia with clinical pictures of keratoacanthoma: A rare case report

“…The etiology and pathogenesis of ALHE is still unknown; however, researcher believed that this entity is formed due to vascular malformation. Some cases of ALHE have been connected into trauma history and Human T‐cell Lymphotropic virus (HTLV) or Human Herpesvirus 8 (HHV‐8) infection …”

Angiolenfoid Hyperplasia with Eosinophilia Treated with Cryotherapy: Case Report

“…Nadiren yaygın lezyonlar görülebilmektedir. 8,9 Bazen derin yumuşak dokuları tutmakta ya da derin damarlardan kaynaklanmaktadır. 7 Serum hipereozinofili ve bölgesel lenfadenomegali gö-rülmektedir, ancak tanı için gerekli değildir.…”

“…Başka bir güncel bildiride skrotum lokalizasyonlu EAH hastasında T-hücre klonalitesi gösterilmiştir. 9 İlginç olarak, iki EAH raporunda birbirini izleyen ya da metasenkron olarak tanı, almış periferal T-hücre lenfoması olması, EAH'nin erken form bir lenfoma olabileceği sorusunu akla getirmiştir. 12,14 Ancak hastaların büyük çoğunluğunun benign karakterde olduğu vurgulanmaktadır.…”

Angiolymphoid hyperplasia with eosinophilia occurring in bilateral eyelids

“…On the other hand, others considered these lesions as neoplastic [7]. Previous papers have reported that these masses are consisted of mostly CD3-positive T cells [8] with monoclonality [9]. …”