title: 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.
authors.affiliation: Cellular Pathology, Hospital El Bierzo, Ponferrada, Spain.|CellCOM-ST Group, Biomedical Investigation Institute of A Coruña, A Coruña, Spain.|Cellular Pathology, Hospital de la Reina, Ponferrada, Spain.
authors.forename: Angel
authors.identifier: http://orcid.org/0000-0001-6617-2364
authors.initials: A
authors.lastname: Fernandez-Flores
authors.affiliation: Department of Pathology, Southern California Kaiser Permanente, Los Angeles Medical Center (LAMC), Kaiser Permanente, Los Angeles, California.
authors.forename: David S
authors.identifier: http://orcid.org/0000-0001-8875-0858
authors.initials: DS
authors.lastname: Cassarino
mesh_terms: D000368:Aged; D000796:Angiolymphoid Hyperplasia with Eosinophilia / Q000150:complications / Q000473:pathology*; D005260:Female; D006801:Humans; D007154:Immune System Diseases / Q000150:complications; D015451:Leukemia, Lymphocytic, Chronic, B-Cell / Q000150:complications; D008297:Male; D008875:Middle Aged; D018294:Neoplasms, Adnexal and Skin Appendage / Q000150:complications
keywords: IgG4-related disease; Kimura disease; angiolymphoid hyperplasia with eosinophilia; chronic lymphocytic leukemia; epithelioid hemangioma; erythema elevatum dyutinum