Angiolymphoid Hyperplasia with Eosinophilia or the Kimur’s Disease?

Overview

Angiolymphoid hyperplasia with eosinophilia (ALHE) and Kimur’s disease aretworarely occurringandmutually very similar, yet independentnosological units.For the frequency of similar features these diseases were long considered to bemodifications of one disease. In the last years most authors considered thesediseases to be independent entities with certain specific clinical and morphologicalfeatures. The case of a 76-year white man, suffering from occurrence ofsubcutaneous tumor formations in various part of the body for the period of sixyears, is described. Six years after the first signs of the disease, swelling of eyelidson the right eye with palpable resistance under the upper temporalmargin of eyesocket developed. A similar finding also developed on the left side eight monthslater. The tumors were removed by surgery and subjected to histological examination.One year later a unilateral relapse developed to be again subjected tosurgical treatment and histological examination. In the first two orbital excisionsthe tumor infiltrate was in topical connection with structure of the tear gland, thethird excision did not contain such structures. The histological diagnosis wasestablished as ALHE in spite of some common features with the Kimur’s disease.The diagnosis of ALHE was especially supported by absence of the formation oflymphatic follicles, absent fibrotization in the infiltrate and epithelium-like or even histiocyte-like appearance of proliferating endothelia, which formed minutevascular lumina. A substantial exceptionality in the occurrence of the Kimur’sdisease in white Europeans further supported the diagnosis of ALHE. Our ownexperience indicates that both units mutually overlap even in features consideredto be of distinctly different character.Aquestion arises,whether the strict divisionof these two nosological units is really possible in practice with unequivocalcertainty.

Key words:
angiolymphoid hyperplasia with eosinophilia, Kimur’s disease, bilateralaffection of the tear gland, relapse