• Title/Summary/Keyword: ALHE

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RECURRED ANGIOLYMPHOID HYPERPLASIA WITH EOSINOPHILIA (호산구증가증을 동반한 혈관임파양 증식증)

  • Byun, Young-Nam;Kim, Jong-Chul;Choung, Pill-Hoon;Yoon, Young-Ho
    • 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.

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THE CASE REPORT OF ANGIOLYMPHOID HYPERPLASIA WITH EOSINOPHILIA ON CHIN (하악 이부에 생긴 Angiolymphoid hyperplasia with eosinophilia(Kimura's disease)의 치험례)

  • Yang, Yun-Seok;Cho, Yong-Seok
    • 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.

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Angiolymphoid hyperplasia with eosinophilia attached to the masseter muscle

  • Kwon, Hyo Jeong;Jung, Ee Room;Choi, Jong Yun;Seo, Bommie Florence;Kwon, Ho;Jung, Sung-No
    • 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.

ANGIOLYMPHOID HYPERPLASIA WITH EOSINOPHILIA : A CASE REPORT (호산구 증가중을 동반한 ANGIOLYMPHOID HYPERPLASIA의 증례보고)

  • Kim, Young-Kyun;Yeo, Hwan-Ho;Lee, Cheol-Woo;Yang, In-Seok;Cho, Se-In;Cho, Jae-O
    • 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).

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Angiolymphoid Hyperplasia with Eosinophilia (ALHE) of the Oral Cavity: a Case Report (구강내에 발생한 혈관림프증식증: 증례보고)

  • Cho, Dong-Baek;Park, Ji-Un;Baek, Ji-Young;Choi, Won-Sik
    • 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.

Angiolymphoid Hyperplasia with Eosinophilia mimicking Parotid Tumor (이하선 부위에 발생한 Angiolymphoid Hyperplasia with Eosinophilia)

  • Kim, Que-Chic;Noh, He-Il;Auo, Hyeon-Jin;Chun, Byung-Joon;Cho, Jung-Hae;Kang, Seok-Jin
    • 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.

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REPORT OF EXPERIENCE WITH KIMURA'S DISEASE (기무라씨 질환, 5 예 보고)

  • Seel David J.;Park Yoon-Kyu;Lee Kwang-Min
    • Korean Journal of Head & Neck Oncology
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    • v.5 no.1
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    • pp.39-46
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    • 1989
  • Kimura's Disease is a chronic inflammatory and proliferative condition producing subcutaneous masses especially in the head and neck area. This report of our experience with 5 patients with this disease is the first in the Korean surgical literature. Kimura's Disease is thought to be part of the larger spectrum of the entity known as angiolymphoid hyperplasia with eosinophilia (ALHE). It is characterized pathologically by hyperplastic lymphoid follicles, eosinophilic infiltration, and vase 비 ar proliferation. It produces masses which are most common in the area of the parotid, submandibular gland and upper neck. These masses occupy the subcutaneous tissues but also extend into salivary tissue and into upper neck nodes. One of our patients had masses in the groin. The tumors are extremely vascular due to the presence of new proliferative vessels and sinusoids. The average age of our 5 patients was 35, but all but one case were younger than 38 years of age. The male: female ratio was 3 : 2, and the average duration of symptoms was 5,2years. All patients had peripheral blood eosinophilia. All had multiple masses, sometimes symmetrical. The management was surgery alone in one case, surgery and steroids in one case, surgery and radiotherapy in two cases, and all three modalities in one case. The relationship of this entity to ALHE and our experience in the management of this disease are presented. A clinicopathological discrepancy alerted us to the existence of Kimura's Disease. A nineteen-year old male presented with subcutaneous masses over both mastoid areas present for 3 years (Case III). When biopsy on each side was reported as 'eosinophilic granuloma' we submitted the slides to an internationally expert pathologist. Symmetrically occurring tumors in the peri-parotid subcutaneous areas did not fit any category of neoplasm or granuloma known to us. The diagnosis, made by Dr. Gist Fan at the Ochsner Clinic, was Kimura's Disease. We found two additional cases in a review of soft tissue eosinophilic granuloma previously reported at Presbyterian Medical Center, and since then have diagnosed two new cases. These five cases constitute the basis for this, the largest series to be reported in Korea. These vascular, tumor-like lesions of the skin, subcutaneous areas and subjacent structures of the head and neck have been a variety of names, such as angiolymphoid hyperplasia with eosinophilia, eosinophilic hyperplastic lymphogranuloma, angioblastic lymphoid hyperplasia with eosinophilia, histioid hemangioma, and epithelioid hemangioma. The history of this disease spectrum dates back to 1937 when Kimm and Szeto (1) reported 7 cases of 'eosinophilic hyperplastic lymphogranuloma' in the Proceedings of the Chinese Medical Journal. In 1948 Kimura and his associates(2) reported additional cases in Japan under the title 'On the unusual granulation combined with hyperplastic changes of lymphatic tissue.' From then until 1966 several hundred cases were reported in China and Japan. The first report from the West was by Wells and Whimster(3) in the British Journal of Dermatology, in 1969. These authors coined the term, angiolymphoid hyperplasia with eosinophilia (ALHE). Since that time a debate has ensued as to whether Kimura's Disease and ALHE are distinct entities, or whether Kimura's is part of the larger spectrum of ALHE, perhaps a later or advanced phase. From the clinical perspective, surgeons should be aware of the diagnosis of Kimura's Disease not only as part of the differential diagnosis of head and neck tumors but also because these lesions are indolent, and generally require conservative surgical removal as part of the management program. CASE I. A 37-year-old female company employee presented in August 1982 with submental swelling of 12 years' duration and with inguinal swelling of 7 years' duration. The submental mass measured 5x5cm. and the inguinal mass was 8x4cm. in size. Peripheral eosinophilia varying from 14% to 40% was found. On August 20, 1982, the submental mass was removed and a superficial groin dissection was done. In May 1983 an intraoral lesion of the palate was removed. The patient is free of disease. CASE II. A 23-year-old unemployed man visited this hospital for the first time in July, 1984, with swelling of the right cheek present for 6 years. The mass was soft and ill-defined but measured 10x20cm. and extended from the submandibular upper neck to the zygomatic arch, and from the mastoid to the cheek, over the parotid gland. Eosinophilia varying from 27% to 29% was noted in the peripheral blood. On March 21, 1986, the lesion was resected. The procedure comprised an extended superficial parotidectomy from the temporalis fascia to the upper neck. Post-operatively radiotherapy 3000 rad tissue dose was administered using the 6 MeV linear accelerator. The patient remains free of disease. CASE III. A 19-year-old student came to the clinic with masses over both mastoid areas, present 3 years. On the right there were two adjacent lesions, one over the mastoid, the other in the upper jugular level of the neck. On the left it was a single mass over the mastoid. Eosinophilia varied from 13 to 32% in the peripheral blood, and 11.6% in the bone marrow. Incisional biopsy revealed 'eosinophilic granuloma' and a trial of predisolone was employed. The mass increased in size so a small dose of radiation (600 rads) was used, with substantial regression,. The lesion on the left was excised and follwed by 1000 rads radiotherapy. Finally recurrent tumor on the right side was removed on November 5, 1985. The patient remains free of disease. CASE N. A 29-year-old local merchant had had swelling of both upper necks since childhood. At the time of his first visit on March 17, 1986, the right submandibular mass measured 5x3.5cm. and the ,right upper neck and parotid tail mass measured 2.5cm. On the left there were masses in the upper neck, the largest of which measured 2.5cm, and of the parotid tail, 2.0cm. in size.(See Fig. 1) Peripheral eosinophilia of 39% was recorded. Left side partial parotidectomy and resection of the upper neck and subdigstric mases was done on May 2, 1986. The mass involving the right parotid tail and upper neck nodes was removed on Angust 7,1986. Postoperatively the patient was placed on prednisolone 30 mg. per day. No definite masses are palpable. CASE V. A 66-year-old housewife informed us, at the time of her first visit in May, 1986, that she had had multiple neck masses since 10 years ago. On the right side there was a 2.5cm. subcutaneous mass of the upper neck, over the upper jugular chain. On the left there was a 9x4.5cm. mass involving the entire parotid, the post-auricular area and the upper neck. A third mass presented in the submental area and measured 3.5cm. (See Fig. 2) Eosinophilia of 51% was noted in the peripheral blood. partial excision of the left upper neck lesion and complete excision of the submental mass were performed on june 6, 1986. post-operatively she was placed on 20 mg. of prednisolone daily, but when the mass re-grew after two months she was referred to Radiation Therapy for a 2500 rad course of treatment. A barely palpable thickening remains.

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A Case of Kimura's disease (경부에 발생한 기무라씨병 1례)

  • 김춘일;김상후;정대건;박용수
    • 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.

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