Three unusual histopathological presentations of angiolymphoid hyperplasia with eosinophilia

Abstract

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a poorly understood benign vasculoproliferative disorder. Histopathologically, the lesions are composed of a dermal or subcutaneous proliferation of blood vessels of varying sizes with plump and bland endothelial cells, which often show vacuolization and protrusion of the cytoplasm into the lumen. The vascular proliferation is admixed with a diffuse inflammatory infiltrate composed of lymphocytes, eosinophils and mast cells. Lymphoid follicles with germinal centers can sometimes be seen. In addition, fibrosis is a common accompanying feature. Some clinical and histopathological variants of ALHE have already been described in the literature. In this report, we present 3 rare associations of ALHE that have not been previously described. Case 1 was a 73-year-old woman with a lesion on her right medial thigh. Examination showed ALHE admixed with a chronic lymphocytic leukemic (CLL) infiltrate. Case 2 was a 55-year-old woman with a lesion on her right anterior ankle, which was a syringocystadenoma papilliferum co-existing with an ALHE. Case 3 was a 54-year-old man with a lesion on the left medial thigh, which showed overlapping features of IgG4-related disease associated with areas of ALHE. Given these multiple and diverse associations, it seems likely that ALHE may be a reactive rather than neoplastic process.

Rare angiolymphoid hyperplasia with eosinophilia examined through fine needle aspiration cytology, histopathology and immunophenotypic characterization: A case report

“…The disease mainly occurs in Asia, with a slightly higher incidence in male patients compared with that in female patients and a wide range of age of onset (8). The tumor, which is mainly distributed in the head and neck, and more specifically around the ear and auricle, is presented as dermal papules or nodules, commonly accompanied by itching and scratching; therefore, the patients are prone to bleeding (9). The early stage of the disease is characterized by vascular proliferative lesions, while the late stage presents significant lymphoproliferative lesions, which needs to be differentiated morphologically from Kimura's disease (8,10).…”

Cutaneous epithelioid haemangiomas show somatic mutations in the mitogen‐activated protein kinase pathway

“…6 When multiple, EHs usually appear grouped and may be confluent. Some clinical and histological findings indicate a reactive nature of EH, 1,7 such as history and evidence of previous inflammatory stimuli or trauma. However, the aetiology and pathogenesis of EH are still unclear.…”

Angiolymphoid hyperplasia with eosinophilia of the tongue with a Sézary syndrome history: Evidence for immune mediation of the disease

“…A recent case report coexisting with peripheral T cell lymphoma explored the relationship to haematologic malignancy, which can exhibit eosinophilic dermatosis. In view of the variety of concomitant disease processes, the authors concluded that angiolymphoid hyperplasia with eosinophilia is more likely to be a reactive phenomenon than a true neoplasm 7 …”

“…In view of the variety of concomitant disease processes, the authors concluded that angiolymphoid hyperplasia with eosinophilia is more likely to be a reactive phenomenon than a true neoplasm. 7 Recently, it has been reported that THSD7A, a protein that causes membranous nephropathy, is upregulated by eosinophils in angiolymphoid hyperplasia with eosinophilia and that there is increased VEGF-A expression, especially in conditions that support the Th type 2 phenotype. The authors hypothesised that immune responses mediated by Th type 2 cells might link angiolymphoid hyperplasia with eosinophilia to membranous nephropathy.…”