• Maxillofacial Plastic and Reconstructive Surgery
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• v.32 no.6
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• pp.573-576
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• 2010

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular lesion that is characterized by proliferation of small to medium-sized vascular structures lined by histiocytoid or epithelioid endothelial cells and often accompanied by an inflammatory infiltrate comprising lymphocytes, plasma cells and eosinophils. A 34-year-old man without any generalized systemic conditions presented with a slowly enlarging painless swelling in the buccal area. An excisional biopsy was conducted and the diagnosis of Angiolymphoid hyperplasia with eosinophilia was confirmed. We report a case of angiolymphoid hyperplasia with eosinophilia of the buccal area in oral cavity and review the previously reported cases and literatures of angiolymphoid hyperplasia with eosinophilia.

• Korean Journal of Bronchoesophagology
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• v.8 no.2
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• pp.51-55
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• 2002

Angiolymphoid hyperplasia with eosinophilia(ALHE) is an uncommon idiopathic condition that presents with isolatedor grouped cutaneous plaques or nodules of the head and neck. Extracutaneous involvement is rare. ALHE is a distinct pathologic entity marked by a proliferation of blood vessels with distinctive large endothelial cells accompanied by a characteristic inflammatory infiltrate that includes eosinophils. The lesion is benign but may be persistent and difficult to eradicate. The authors have recently experiecned a case of angiolymphoid hyperplasia with eosinophilia in a 52-year-old male who had a painless enlarging mass in his right preauricular area and external ear canal for several years. We present this case with the review of literatures.

• Maxillofacial Plastic and Reconstructive Surgery
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• v.15 no.1
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• pp.27-34
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• 1993

Angiolymphoid hyperplasia with eosinophilia is an unusual and controversial lesion that occurs primarily in the head and neck area. This lesion was usually confused with Kimura's disease. We present the case of a 32-year-old woman with massive soft movable mass in left facial area which was diagnosed preoperatively as a fasciitis nodular. The final histologic diagnosis of the excised mass was angiolymphoid hyperplasia with eosinophilia(ALHE).

• Maxillofacial Plastic and Reconstructive Surgery
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• v.18 no.3
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• pp.443-447
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• 1996

Angiolymphoid hyperplasia with eosinophilia(ALHE) is an uncommon, benign vascular proliferation with the background of a stroma which is heavily infiltrated by lymphocytes and eosinophils and includes lymphatic follicles with prominent germinal centers. ALHE primarily involves the skin and subcutis in head and neck as various forms of nodules. There has been considerable controversy about the relationship between ALHE and Kimura's disease. Kimura's disease, originally reported by Kimura et el., is an unusual granulation with proliferation of lymphoid tissue. Wells and Whimster published the first report describing a condition that resembled Kimura's disease and designated it as ALHE. For a time being two lesions are thought to be same lesion, but recently they are considered as two different entities, histopathologically. The cause of this disease remains unknown, and physicians have used a variety of treatment modalities including cryosurgery, steriod therapy, electrodesiccation, curettage, radiotherapy, laser therapy and surgical excision. But any treatment modality leaves problem of recurrence because the lesion is not well encapsulated. Being poorly encapsulated, the lesion's remnants are apt to be left and this markes some problems : recurrence and possible adjacent organ injury. In this case we misdiagnosed the lesion as well encapsulated benign mass. We performed excisional biopsy and experienced prolonged operation time and unwanted mentalis muscle injury. We think that the importance of poor encapsulation of ALHE should be stressed. So we report our experience with literature review.

• Archives of Craniofacial Surgery
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• v.25 no.4
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• pp.192-196
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• 2024

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare condition characterized by nodular lesions predominantly in the head and neck region, often causing discomfort or pain. Treatment remains challenging because of its rarity and the lack of established guidelines. This report presents a case of ALHE affecting the earlobes that was successfully managed using ear lobule reduction surgery and subsequent intralesional steroid injections. A 31-year-old woman with a history of recurrent earlobe masses underwent a partial excision to avoid the loss of the earlobe. Histopathological examination confirmed Kimura disease, a variant of ALHE. Subsequent local methylprednisolone injections effectively controlled the remaining lesions, resulting in significant size reduction without notching. Various treatment modalities have been attempted for this condition; however, recurrence rates remain high. Surgical resection combined with intralesional corticosteroid injections is the preferred approach. In this case, a sub-antitragal groove technique for earlobe reduction was employed to preserve the lateral edge of the ear lobule, minimize the risk of deformity, and achieve a predictable outcome. The sub-antitragal groove technique offers an approach to reduce earlobe size without compromising aesthetics. Further research is required to elucidate the pathogenesis of ALHE and establish standardized treatment protocols for this rare condition.

• Archives of Craniofacial Surgery
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• v.21 no.5
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• pp.319-322
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• 2020

Angiolymphoid hyperplasia with eosinophilia (ALHE) is a rare benign vascular tumor. The pathogenesis of ALHE is unknown; however, it may be linked to local trauma. ALHE predominantly occurs in areas of the preauricular region, forehead, and scalp; the masseter area is rarely involved. A 49-year-old man was referred for a mass in the right cheek region that was felt 2 months prior. Physical and imaging examination results suggested the presence of a benign tumor. Thus, surgical excision was performed. Pathologic findings confirmed an unexpected diagnosis of ALHE. This case was interesting, since the mass occurred at an unusual site with a misdiagnosis of an epidermal inclusion cyst.

• Korean Journal of Head & Neck Oncology
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• v.29 no.1
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• pp.22-25
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• 2013

Angiolymphoid hyperplasia with eosinophilia(Kimura's disease) is a relatively uncommon chronic benign disorder of unknown etiology, predominantly in the head and neck region and associated with lymphadenopathy or salivary gland enlargement. The treatment options are still controversial including medical therapy, surgery, and radiation. Kimura's disease of bilateral postauricular involvement has rarely been reported. We report a male patient with sequentially developed Kimura's disease involving the bilateral postauricular region and was treated by combined surgical excision and steroid therapy.

• Korean Journal of Bronchoesophagology
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• v.3 no.2
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• pp.323-326
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• 1997

Kimura's disease is a chronic inflammatory disease which often presents as a tumor like swelling in the head and neck region. This lesion is benign but it may easily be mistaken for a malignant tumor. Kimura's disease has been confused with angiolymphoid hyperplasia with eosinophilia(ALHE). The cause is unknown, but theories include autoimmune, allergic, neoplastic, and infectious cause by insect bites and parasites. The treatment of choice is surgical removal. Recently we experienced a case of Kimura's disease occuring in the both side of posterior neck triangle area of a 37 year old male patient who was treated with surgical excision and systemic corticosteroid administration.

• Maxillofacial Plastic and Reconstructive Surgery
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• v.18 no.4
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• pp.636-646
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• 1996

Angiolymphoid hyperplasia with eosinophilia(ALHE) is a benign subcutaneous lesion that primarily affects head and neck region. It is characterized by single or multiple nodules in the subcutaneous tissue associated with eosinophilia in the peripheral blood. Kimura's disease, originally reported by Kimura et al., is similar lesion with ALHE in the clinical and histopathological aspects. There has been considerable controversy about the relation between Kimura's disease and ALHE. In Korea, 26 cases of Kimura's disease and ALHE have been reported since 1975. We present a case of recurred ALHE occurring on the left cheek in a 58-year-old woman. Including this case, we summarized all cases that were reported as Kimura's disease or ALHE in Korea through literature review. We also give an outline of clinical and histopathological characteristics of cases reported in Korea.

• Journal of Yeungnam Medical Science
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• v.30 no.2
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• pp.149-151
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• 2013

Kimura's disease is an angiolymphoid-proliferative disorder that manifests with benign subcutaneous swelling predominantly in the head and the neck. Kidney involvement, including proteinuria, occurs in 12-16% of patients with the disease, and 60-78% of such cases is nephrotic syndrome. Reported etiologies of nephrotic syndrome in Kimura's disease include membranous glomerulonephritis, mesangial proliferative glomerulonephritis, minimal-change disease, focal segmental glomerulosclerosis, diffuse proliferative glomerulonephritis and immunoglobulin A (IgA) nephropathy. There have been only two case reports of IgA nephropathy in Kimura's disease, in 1998. In this report, we present a third case of IgA nephropathy associated with Kimura's disease.