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1/13. Localized soft tissue angiomatosis with subsequent development of angiolymphoid hyperplasia with eosinophilia.

    We report an unusual case of arteriovenous (AV) malformation (localized form of soft tissue angiomatosis): a reaction of angiolymphoid hyperplasia with eosinophilia (ALHE) developed over the lesion of this pre-existing AV malformation. To the best of our knowledge, there is no other report of ALHE in association with a pre-existing bona fide AV malformation. A 71-year-old woman consulted our clinic because a reddish, dome-shaped tumor on her preauricular region, present as a slightly elevated, skin-colored tumor since her childhood, had rapidly enlarged and become more elevated with color change during the preceding two months. The histological features of most of the lesion corresponded to those of soft tissue angiomatosis (localized form). The proliferative vessels underlying the soft tissue angiomatosis streamed out and welled into the dermis with an accompanying prominent inflammatory infiltrate composed of lymphoid cells and eosinophils: these histological features were characteristic of ALHE. The recent abrupt clinical changes in the tumor were considered to represent a reaction to the angiomatosis with development of ALHE.
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2/13. Detection of Epstein-Barr virus dna in a patient with Kimura's disease.

    An 80-year-old man, with a past medical history of senile dementia, presented with a 6-month history of a solitary, gradually enlarging tumor, located on his chin. A squamous cell carcinoma had been surgically excised 30 years previously in the same location. physical examination revealed an erythematous, well-defined plaque of 3 cm in diameter, located on the chin (Fig. 1). The submandibular lymph nodes were enlarged. Squamous cell carcinoma and primary cutaneous lymphoma were considered. Relevant laboratory findings were as follows: white blood cell count, 5.600/microL; eosinophils, 1000/microL; gammaglobulin, 2.4 g/dL; lactate dehydrogenase, 343 IU/L; and immunoglobulin g (IgG) antibodies to Epstein-Barr virus (EBV) positive (at 1 : 128 serum dilution), with negative IgM. skin and lymph node biopsies were performed. Histopathologic study of the cutaneous specimen revealed a heavy lymphoid infiltrate with numerous lymphoid follicles, with prominent germinal centers involving the subcutaneous fat as well as the deep dermis and muscular fascia. Some germinal centers showed folliculolysis. The lymphoid follicles were surrounded by fibrous tissue. The interfollicular infiltrate was rich in plasma cells and eosinophils that formed scattered eosinophilic microabscesses. Thin-walled vessels were numerous and prominent, but with no epithelioid or vacuolated endothelial cells (Fig. 2). Histopathology of a lymph node biopsy specimen showed reactive lymphoid follicle hyperplasia, with prominent eosinophilic infiltrates in both follicular and interfollicular areas. Eosinophilic deposits and polykaryocytes of Warthin-Finkeldey type were seen in the germinal centers. The paracortical area showed vascular proliferation. polymerase chain reaction (PCR) for the detection of specific sequences of EBV from routinely processed paraffin-embedded material was carried out under the conditions and with the same set of primers as described previously in detail (Tenorio A, Echevarria JE, Casas E et al. J Virol methods 1993; 44: 261-269). dna samples were confirmed to be amplifiable with PCR primers specific for a conserved region of the human beta-globin gene. Every sample was tested at least twice for EBV dna and beta-globin gene. One sample from one skin lesion of the patient, with confirmed diagnosis of Kimura's disease, and 10 samples from normal skin biopsies retrospectively collected from other patients in archival files of our department were tested. Only the patient's specimen tested positive to EBV. The amplified product of EBV was analyzed using dna sequencing and confirmed the results obtained. The patient received radiotherapy at doses of 35 Gy. Nevertheless, the tumor enlarged to reach twofold its original size 1 month later. Due to the physical status of the patient, no further treatments were considered, but the disease remained stable over the following 3 years.
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3/13. angiolymphoid hyperplasia with eosinophilia of the tongue: report of a case and review of the literature.

    angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon disorder manifested as solitary or multiple nodules in dermis and subcutaneous tissue of head and neck region. ALHE affecting tongue is very rare. We report a 56-year-old man with ALHE of the tongue and review the previously reported cases of ALHE of the tongue.
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4/13. Juvenile temporal arteritis is a manifestation of Kimura disease.

    An asymptomatic nodule appeared in the right temporal region of an 81-year-old woman. Histopathologic examination confirmed significant thickening of the vascular wall, constriction of the vessel lumen, and infiltration of numerous eosinophils and lymphocytes. giant cells were not seen. Lymphoid follicles and capillaries surrounded the large vessel. Elastica van Gieson staining revealed a laceration of the internal elastic lamina. Based on these clinical and histologic findings, the patient was diagnosed as having juvenile temporal arteritis (JTA), a disease first proposed by Lie and his colleagues in 1975. Three years later, a new eruption, again asymptomatic, appeared in the posterior region of the patient's right ear. Subsequently, she was referred to our department. Histologic examination of the new lesion confirmed the infiltration of lymphocytes and eosinophils, which was accompanied by numerous lymphoid follicles, and the proliferation of endothelial cells and capillaries from the deep dermis to the subcutaneous tissue. The patient was diagnosed as having Kimura disease, which is a persistent and recurrent illness. We hypothesized that JTA was a partial expression of Kimura disease and investigated whether past cases of JTA could be considered Kimura disease. As a result, we found that most cases of JTA could indeed be considered Kimura disease. Furthermore, we examined the vascular changes in the routinely and elastic fiber-stained sections of three cases with Kimura disease and two cases with angiolymphoid hyperplasia with eosinophilia. The results showed occlusive vascular changes in most samples from these cases, supporting the hypothesis that JTA is an accessory lesion of Kimura disease.
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5/13. Protruding tumorous angiolymphoid hyperplasia with eosinophilia (ALHE) of the scalp accompanied by arterial occlusion.

    We report a case of an extraordinarily large tumorous form of ALHE developing on the occiput of a 57-year-old Japanese male. Histologically, it was characterized by increased numbers of small blood vessels, fibrosis, and lymph follicle formations with massive eosinopilia in the dermis in addition to an occluded artery in the deep dermis.
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6/13. A case of eosinophilic lymphfolliculosis of the skin (Kimura's disease).

    Eosinophilic lymphfolliculosis of the skin (Kimura's disease) in a 31-year-old male is reported. He had two subcutaneous tumors, 10 x 9 x 3 cm and 9 x 7 x 2 cm in size, in the right retroauricular and submaxillary areas respectively. Histopathologic examination of the tumors showed marked infiltration of eosinophils, mast cells and lymphocytes, and angiomatous proliferation of blood vessels from the dermis to the skeletal muscular tissue. In subcutaneous adipose tissue, there were numerous lymphfollicles with germinal centers in which IgE deposition with reticular appearance was observed by immunofluorescent technique. Study of the peripheral blood showed marked eosinophilia and an increase of serum IgE level. These findings suggest the possibility that Type 1 allergic reaction may be involved in the histogenesis of eosinophilic lymphfolliculosis of the skin, although there is no direct evidence for the existence of antigens which react to IgE antibody in the skin lesion.
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7/13. Pseudopyogenic granuloma: effects of corticosteroid on newly-formed vessels.

    Papular lesions of pseudopyogenic granuloma were examined before and during treatment with systemic corticosteroid using both light and electron microscopy. Before treatment, these lesions showed a proliferation of vessels lined with plump endothelial cells and heavy lymphohistiocytic infiltrates with numerous eosinophils in the dermis. During treatment, flattened papules showed a dilatation of vessels lined with flattened endothelial cells, extravasation of red cells, discontinuity of endothelial basal lamina, and, ultimately, discontinuity of the endothelial cells themselves. eosinophils were rarely found. From these findings, it can be said that the morphological effects of corticosteroid on microvasculature have in part been elucidated.
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8/13. angiolymphoid hyperplasia with eosinophilia of the tongue: report of a case and review of the literature.

    angiolymphoid hyperplasia with eosinophilia has been described mainly in the dermis and subcutaneous tissues of the head and neck, and only occasionally in the oral cavity. A case of angiolymphoid hyperplasia with eosinophilia of the tongue in an 82-year-old man with typical morphologic features is presented. The diagnosis criteria and treatment of this lesion reported in the English literature are also reviewed.
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9/13. angiolymphoid hyperplasia with eosinophilia (Kimura's disease): report of a large-sized lesion.

    angiolymphoid hyperplasia with eosinophilia (ALHE) is an uncommon lesion with a predilection for skin of the head and neck region. It usually presents as one or a few smooth-surfaced nodules which are often asymptomatic. Histologically the nodules reveal two components: abnormal proliferating vessels and a cellular infiltrate consisting of lymphocytes and eosinophils, involving both the dermis and the subcutaneous tissue. A case is presented of an unusually large recurrent lesion on the scalp.
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10/13. angiolymphoid hyperplasia with eosinophilia associated with tetanus toxoid vaccination.

    Three cases of angiolymphoid hyperplasia with eosinophilia are reported at the site of tetanus toxoid vaccination. All presented as nodules on the upper arm, a known adverse effect of the vaccine following superficial inoculation. Histologically the nodules showed numerous small and medium sized vessels, some lined by hyperplastic endothelial cells with perivascular lymphocytic cuffing and an inflammatory infiltrate of plasma cells, eosinophils and scattered mast cells. Lymphoid follicles replete with germinal centres were identified in the dermis and subcutaneous tissue. Immunostaining revealed a polytypic lymphoplasmacytic infiltrate and a curious IgE reticulated pattern within the germinal centres, a feature described in both Kimura's disease and angiolymphoid hyperplasia. The present study suggests that atopic reaction to tetanus toxoid may be an aetiological factor in some cases of angiolymphoid hyperplasia.
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