Angiolymphoid hyperplasia with eosinophilia: A disease that may be confused with malignancy
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Twelve new cases of an unusual, benign vasoproliferative and inflammatory disorder of unknown etiology, angiolymphoid hyperplasia with eosinophilia (ALHE), are described and contrasted clinically and pathologically with those appearing in the literature. Only recently recognized in the United States, the disease is of singular importance because the vascular component may be confused histologically with angiosarcoma, thereby resulting in unwarranted aggressive therapeutic measures. ALHE characteristically affects adults and presents in the head and neck region as either solitary or multiple cutaneous tumors. The lesions are pruritic, frequently bleed after minor trauma, and may be associated with peripheral eosinophilia and regional lymphadenopathy. On rare occasions, the disease may masquerade as a salivary-gland tumor, cause stenosis of the external auditory canal, or present as an osseous lesion of the skull. Extrafacial tumors are uncommon. Excision is the most frequent form of therapy; however, local irradiation, corticosteroids, electrodessication with curettage, and chemotherapy have also had varying degrees of success.Keywords:
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Abstract Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign condition affecting principally the head and neck region of young females. We describe a 42‐year‐old female patient of ALHE showing the typical changes of endothelial cells and features similar to Kimura's disease in histologic and immunohistochemical findings.
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Abstract Background Much controversy has existed with regard to the relationship between Kimura's disease and angiolymphoid hyperplasia with eosinophilia. They were initially thought to represent the same disease spectrum, but it has now been widely accepted that they are two separate disease entities. To our knowledge, there have been no reports to date describing a case of both Kimura's disease and angiolymphoid hyperplasia with eosinophilia coexisting in the same patient. Methods We describe a patient presenting initially with a right postauricular subcutaneous swelling and subsequently developing multiple erythematous facial papules and nodules. The medical literature is reviewed. Results Clinical features of the right postauricular subcutaneous swelling and multiple erythematous facial papules/nodules suggest Kimura's disease in the former and angiolymphoid hyperplasia with eosinophilia in the latter. Histopathological examinations of these lesions helped to confirm the diagnosis of Kimura's disease and angiolymphoid hyperplasia with eosinophilia, respectively. Conclusions Kimura's disease and angiolymphoid hyperplasia with eosinophilia can coexist in the same patient. Coexistence of the two types of lesions in one patient may also be considered evidence that Kimura's disease and angiolymphoid hyperplasia with eosinophilia form a spectrum in one disease.
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Kimura's disease
Lymphoid hyperplasia
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Eosinophils induce tissue damage and organ dysfunction mainly through antigen-antibody reactions, release of toxic granules, mitochondrial DNA, and so on. Skin lesions induced by eosinophilia include erythema, plaques and nodules, usually with itching. Eosinophilia may manifest as an increase of eosinophil count in peripheral blood and eosinophilic infiltration of organs and tissues. Diseases associated with eosinophilia in both peripheral blood and the skin include hypereosinophilic syndrome, Churg-Strauss syndrome, Kimura′s disease, Wells′ syndrome, etc. Dermatoses associated with eosinophilia in peripheral blood alone include allergic cutaneous vasculitis, drug reaction with eosinophilia and systemic symptoms, etc, and those associated with eosinophilia in the skin alone include angiolymphoid hyperplasia with eosinophilia, pemphigoid gestationis, incontinentia pigmenti, etc.
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Eosinophilia; Skin diseases; Churg-Strauss syndrome; Inflammation; Immunity
Incontinentia Pigmenti
Hypereosinophilic Syndrome
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We report a case of angiolymphoid hyperplasia with eosinophilia (ALHE) and nephrotic syndrome. The clinical and histologic features of ALHE are described and contrasted with Kimura's disease. Both ALHE and Kimura's disease may be associated with renal disease.
Kimura's disease
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The clinico-pathological differences between Kimura's angiolymphoid hyperplasia with eosinophilia are presented to emphasize that they are two separate disease entities which until now were considered as the same disease.
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Peripheral blood eosinophilia represents a frequent finding in routine clinical practice when absolute eosinophil count is found to be greater than 0.5 x 109/L (500/µL). Common causes include parasitic infections, allergic reactions, and hyper-eosinophilic syndrome. Eosinophilia secondary to malignancy represents an uncommon presentation. Here we report an atypical case of a 47-year-old previously healthy male who presented to a primary care setting complaining of fatigue and anorexia for the last two weeks. The evaluation revealed leucocytosis and peripheral hypereosinophilia with an absolute eosinophil count of 14.13×109/L (37%). Following an extensive diagnostic work in a secondary care centre he was finally diagnosed with rectal carcinoma. This case highlights that solid malignancy should be considered in patients with marked peripheral eosinophilia.
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Abstract Angiolymphoid hyperplasia with eosinophilia ( ALHE ), also known as epithelioid hemangioma, is a benign vascular proliferation of uncertain etiology. Herein, we present the first documented case of ALHE following venipuncture and review the published work regarding the relationship of this entity to preceding trauma.
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Until now, there have only been two cases describing the coexistence of angiolymphoid hyperplasia with eosinophilia (ALHE) and Kimura's disease in one patient. Herein, we report the previous case of recurrent ALHE and Kimura's disease that was successfully treated with surgery.
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A case of angiolymphoid hyperplasia with eosinophilia, presenting as a subcutaneous mass over the face in a 25-years-old woman is presented. The lesion was treated by surgical excision. There was no recurrence after a follow up of 2 months.
Kimura's disease
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