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[伴嗜酸性粒细胞增多的血管淋巴样增生累及双侧泪腺]

[Angiolymphoid hyperplasia with eosinophilia with bilateral involvement of the lacrimal glands].

作者信息

Baráková Drahomíra, Sâch Josef, Kuchynka Pavel, Rêdinová Magdalena, Kocur Ivo

机构信息

Die Augenklinik der 3. Medizinischen Fakultät der Karlsuniversität und des Fakultätskrankenhauses Královské Vinohrady, Prague, Czech Republic.

出版信息

Klin Monbl Augenheilkd. 2002 May;219(5):376-9. doi: 10.1055/s-2002-32639.

DOI:10.1055/s-2002-32639
PMID:12094322
Abstract

BACKGROUND

Angiolymphoid hyperplasia with eosinophilia (ALHE) and Kimura's disease are two rarely occurring disorders very similar to each other; however, they are individual nosological entities. For a long time they were considered as a single disease due to the frequency of similar characteristics. The majority of authors have recently considered these diseases as two individual entities with some reciprocal specifications, both clinical as well as morphological.

PATIENT

We report on the case of a 76-year old male white European who had suffered for more than six years from subcutaneous tumour formations in different parts of the body. Eyelid oedema on the right side with palpable resistance under the upper temporal edge of the orbit initially occurred five years after the first symptoms of the disease. A similar finding occurred on the left side after eight months. The tumours on both sides were surgically removed and sent for histological analysis. A unilateral recurrence of the finding appeared after one year, followed by surgical intervention and histological examination.

RESULTS

Tumour infiltrate in the first two orbita excisions was topically related to lacrimal gland structures; these structures were not found in the third excision. The case was histologically diagnosed as ALHE despite some similar characteristics with Kimura's disease. The diagnosis of ALHE was histologically supported, especially by the absence of lymphatic follicle formations and fibrotisation in the infiltrate, and the identification of the appearance of epitheloid up to histiocytoid of proliferating endothelium, which forms small lumens. The diagnosis of ALHE was additionally supported by the fact that the incidence of Kimura's disease in white Europeans is very rare.

CONCLUSIONS

According to our findings, both entities overlap one another, even in some characteristics considered to be distinguishing. The question arises, therefore, whether the strict separation of these nosological entities can indeed be determined with one hundred percent accuracy.

摘要

背景

嗜酸性粒细胞增多性血管淋巴样增生(ALHE)和木村病是两种极为相似的罕见疾病;然而,它们是独立的病种。长期以来,由于具有相似特征的频率较高,它们被视为单一疾病。最近,大多数作者认为这两种疾病是具有一些相互特征的独立实体,包括临床和形态学特征。

患者

我们报告一例76岁的欧洲白人男性病例,他身体不同部位出现皮下肿瘤形成已超过六年。疾病首发症状出现五年后,右侧眼睑水肿,在眶上颞缘下方可触及阻力。八个月后左侧出现类似表现。两侧肿瘤均通过手术切除并送去进行组织学分析。一年后出现该发现的单侧复发,随后进行了手术干预和组织学检查。

结果

前两次眼眶切除术的肿瘤浸润局部与泪腺结构有关;第三次切除术中未发现这些结构。尽管该病例与木村病有一些相似特征,但组织学诊断为ALHE。ALHE的诊断在组织学上得到支持,特别是浸润中无淋巴滤泡形成和纤维化,以及识别出增殖内皮细胞从上皮样到组织细胞样的外观,形成小腔隙。此外,白人欧洲人中木村病的发病率非常低这一事实也支持了ALHE的诊断。

结论

根据我们的发现,这两种实体相互重叠,甚至在一些被认为具有区别性的特征方面也是如此。因此,问题在于这些病种实体的严格区分是否真的能以百分之百的准确性确定。

相似文献

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[Angiolymphoid hyperplasia with eosinophilia with bilateral involvement of the lacrimal glands].[伴嗜酸性粒细胞增多的血管淋巴样增生累及双侧泪腺]
Klin Monbl Augenheilkd. 2002 May;219(5):376-9. doi: 10.1055/s-2002-32639.
2
[Angiolymphoid hyperplasia with eosinophilia or Kimura disease?].
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Angiolymphoid hyperplasia with eosinophilia of the lacrimal gland: a case report.泪腺嗜酸性粒细胞增多性血管淋巴样增生:一例报告
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Kimura's disease. Involvement of regional lymph nodes and distinction from angiolymphoid hyperplasia with eosinophilia.木村病。区域淋巴结受累及与嗜酸性粒细胞增多性血管淋巴样增生的鉴别。
Am J Surg Pathol. 1988 Nov;12(11):843-54.
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Kimura's disease of the lacrimal gland mimicking IgG4-related orbital disease.酷似IgG4相关性眼眶疾病的泪腺木村病。
BMC Ophthalmol. 2014 Dec 15;14:158. doi: 10.1186/1471-2415-14-158.
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Angiolymphoid hyperplasia with eosinophilia (alhe): a rare entity for otolaryngologist.嗜酸性粒细胞增多性血管淋巴样增生(ALHE):耳鼻喉科医生眼中的罕见病症。
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引用本文的文献

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Angiolymphoid hyperplasia with eosinophilia: a case series and literature review.血管淋巴样增生伴嗜酸性粒细胞增多症:病例系列及文献复习。
Int Ophthalmol. 2023 Jul;43(7):2457-2467. doi: 10.1007/s10792-023-02644-y. Epub 2023 Mar 4.