Angiolymphoid hyperplasia with eosinophilia should not be confused with Kimura’s disease

Abstract

ABSTRACT.Purpose: To emphasize the differences between angiolymphoid hyperplasia with eosinophilia (ALHE) and Kimura's disease (KD), two entities often confused in the ophthalmic literature. Methods: Case report of a subcutaneous mass in the periocular region of a 45-year old woman and a MEDLINE review of the ophthalmic and non-ophthalmic literature on ALHE and KD. Results: The clinical and histopathological findings of the present case were consistent with ALHE, but not with KD. A survey of the current ophthalmic literature indicates that these two designations are still often used synonymously, despite that non-ophthalmic papers now separate ALHE from KD. Conclusion:The clinical and histopathological features of ALHE are most often distinctly different from KD and these entities should be clearly separated in the ophthalmic literature. Because of the presumed neoplastic origin ALHE has alternatively been termed epitheloid haemangioma and histiocytic haemangioma. Conversely, Kimura's disease (KD) most probably represents an allergic or autoimmune response preferentially occurring in the head and neck region of young Asian males. Because ALHE and KD share many clinical and histopathological features these lesions have repeatedly been confused and the designations used interchangeably. Overwhelming data now indicate that significant differences exist and in general pathology these entities are separated (Urabe et al. 1987;Chan et al. 1989), but in ophthalmology the original, unifying concept has largely remained unchanged. Even authoritative ophthalmic textbooks currently report ALHE and KD as being one single entity (Rubin & Jakobiec 1994;Jakobiec et al. 1996). Only very recently were two cases of periocular KD reported with the purpose to separate this entity from ALHE (Buggage et al. 1999). Herein we present a case of periocular ALHE with features clearly distinguishable from KD in further support of the concept that KD and ALHE are two separate entities. Case ReportA 45-year old Caucasian woman presented with a history of a slowly growing mass over the previous 6-8 months. Examination revealed a palpable mass located immediately superior to the medial part of the left eyebrow. Surgery was performed and a well-circumscribed subcutaneous mass was found. Following complete excision, the specimen was fixed in 4% formaldehyde and submitted for histopathological examination. There have been no signs of recurrence after a follow-up period of 18 months.The mass measured 11¿10¿7 mm in size and was cut in two halves. The cut surface disclosed a central, softer core surrounded by a rim of denser tissue. Both halves were paraffin embedded and routinely processed for light microscopy. Briefly, sections were cut at 4 mm and stained with haematoxylin and eosin, the van Gieson stain for collagen, the periodic acid-Schiff stain and Alcian blue. In addition, sections were immunostained for the CD45 (clone OPD4; product code M0742) and CD20 (clone B-Ly1; product code M0774) markers (Dako, Carpinteria, CA, USA) using the standar...

Kimura’s Disease: A Diagnostic Challenge

“…18 Histologically, the presence of inflammation around medium-sized arteries or veins and evidence of vascular damage (florid fibrointimal proliferation and cuboidal to dome-shaped endothelial cells) are key features in differentiating ALHE from Kimura's disease. 18,20 Additional prominent morphologic features present in our case and characteristic of Kimura's disease include the noncircumscribed inflammatory infiltrates within extranodal subcutaneous tissues and skeletal muscles associated with reactive germinal center formation and eosinophilic microabscesses. Blood vessels, although prominent, resemble those of the high endothelial venules of lymph nodes.…”

“…18,20 ALHE is a rare but distinctive vascular tumor typically presenting in women during early to mid-adult life. 21 Lymphadenopathy is uncommon, and blood eosinophilia is noted in Ͻ10% of cases.…”

MR imaging of cheek lesions of Kimura’s disease: Focusing on the signal abnormalities and the relationship with superficial musculoaponeurotic systems

“…The major differential diagnosis on MR imaging studies is benign/malignant minor parotid tumor, malignant tumors arising in cutaneous or subcutaneous tissue, tuberculous infection, and angiolymphoid hyperplasia with eosinophilia (ALHE) [17]. The minor salivary gland tumors are usually encapsulated or pseudoencapsulated and are limited to the gland, while KD is frequently associated with irregular extension into the subcutaneous tissue.…”

Hiperplasia angiolinfoidea con eosinofilia: Presentación de un caso en cavidad oral y revisión de la literatura

“…La edad y el sexo de la paciente coinciden más con ALHE, además, la paciente no presentaba ni compromiso ganglionar regional ni patología renal, situación más relacionada con EK. La extensión hacia planos musculares, la gran proliferación vascular y la presencia de células endoteliales altas observada histológicamente en este caso, es característica de la ALHE 1,7,13 .…”

“…Aceptado el 16 de agosto, 2006. 1 Centro de Referencia en Salud Cordillera, Santiago de Chile. 2 Servicio de Anatomía Patológica del Hospital Santiago Oriente «Luis Tisné Brousse».…”