Comparison between Kimura's disease and angiolymphoid hyperplasia with eosinophilia: case reports and literature review

Abstract

Kimura's disease (KD) is a rare chronic inflammatory or allergic disease. Angiolymphoid hyperplasia with eosinophilia (ALHE) is a benign vascular neoplasm. Their relationship has always been debated. This article reports two rare cases, one of each disease. One patient was a 48-year-old female that presented with a mass on her right mandible. She also had oedema erythema and wheals on her lower limbs. She was diagnosed with Kimura's disease complicated with chronic urticaria. The second patient was a 23-year-old female that presented with multiple nodules of unequal size on the scalp. She was diagnosed with angiolymphoid hyperplasia with eosinophilia. The first patient recovered after being treated with surgical resection, glucocorticosteroids, cyclophosphamide and radiotherapy. The second patient underwent the first stage of surgical excision and is currently being followed-up. Comparison of the clinical and histopathological features of these two cases supports the theory that KD and ALHE are two separate disease entities.

Keywords: Kimura’s disease; angiolymphoid hyperplasia with eosinophilia; clinical; histopathological.

Figure 1.

Clinical features of case 1, a 48-year-old Chinese female that presented with a progressive mass on her right mandible that was accompanied by oedema erythema and wheals mainly on the lower limbs: (a) a palpable mass that was nontender and non-erythematous, was observed in the right submandibular region; (b) wheals were scattered on her lower limbs; (c) enhanced computed tomography imaging of her nasopharynx showed a soft tissue mass (1.9 cm × 1.1 cm) in the lateral part of the right submandibular gland.

Figure 2.

Histopathological features of case 1, a 48-year-old Chinese female that presented with a progressive mass on her right mandible that was accompanied by oedema erythema and wheals mainly on the lower limbs: (a) preserved lymph-node architectures with hyperplastic lymphoid follicles and patchy expansion of germinal centres (haematoxylin and eosin, original magnification × 5); (b) the interfollicular region contained numerous eosinophils and interstitial fibrosis (haematoxylin and eosin, original magnification × 10); (c) eosinophilic abscesses were observed in some areas (haematoxylin and eosin, original magnification × 40). The colour version of this figure is available at: http://imr.sagepub.com.

Figure 3.

Clinical features of case 2, a 23-year-old Chinese female that presented with multiple nodules on the scalp without pain and itching: (a) there were numerous skin coloured nodules on the occipital area; (b) some of the nodules were approximately the size of a cherry.

Figure 4.

Histopathological features of case 2, a 23-year-old Chinese female that presented with multiple nodules on the scalp without pain and itching: (a) many irregular hyperplastic vessels with hypertrophic endothelium in the dermis. Endothelial cells were protruding into the lumen assuming a spiked appearance (haematoxylin and eosin, original magnification × 100); (b) the vessels were surrounded by a density of lymphocytes and a small number of eosinophils (haematoxylin and eosin, original magnification × 400). The colour version of this figure is available at: http://imr.sagepub.com.