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无外周嗜酸性粒细胞增多的木村病:1例影像学表现类似静脉畸形的罕见且具有挑战性的病例——病例报告及文献复习

Kimura's Disease without Peripheral Eosinophilia: An Unusual and Challenging Case Simulating Venous Malformation on Imaging Studies-Case Report and Review of literature.

作者信息

Dokania Vivek, Patil Digvijay, Agarwal Ketan, Thakur Prajakta, Prajapati Piyush

机构信息

Resident, Department of Ear, Nose and Throat, Krishna Institute of Medical Sciences University, Karad, Maharashtra, India.

Professor, Department of Oncosurgery, Krishna Institute of Medical Sciences University, Karad, Maharashtra, India.

出版信息

J Clin Diagn Res. 2017 Jun;11(6):ME01-ME04. doi: 10.7860/JCDR/2017/28603.10063. Epub 2017 Jun 1.

Abstract

Kimura's Disease (KD) is a rare chronic inflammatory disorder presenting as multiple painless solitary subcutaneous nodules, predominantly in the head and neck region and frequently associated with regional lymphadenopathy and/or salivary gland involvement. Because of painless nature and indolent course, there is usually a delay in the patient's presentation. KD may radiologically mimic other chronic inflammatory conditions like tuberculosis, vascular malformations and neoplasms. Clinical correlation and histological evaluation along with elevated peripheral eosinophil and serum IgE level are considered important for confirmatory diagnosis. We report a case of painless swelling over right submandibular region extending to the right superficial parotid. The haematological reports were within normal limits. Ultrasound (USG), Magnetic Resonance Imaging (MRI) and Magnetic Resonance Angiogram (MRA) favoured a diagnosis of venous malformation. However, histopathological examination of excised lesion confirmed a diagnosis of KD. This case proves the possibility of the KD even in the absence of peripheral eosinophilia and/ or elevated serum IgE level, and may mimic venous malformation on imaging studies. Therefore, KD must find a place in the differentials of solitary painless neck swelling even in the absence of peripheral eosinophilia and/or elevated IgE level.

摘要

木村病(KD)是一种罕见的慢性炎症性疾病,表现为多个无痛性孤立性皮下结节,主要位于头颈部区域,常伴有区域淋巴结肿大和/或唾液腺受累。由于其无痛性和病程缓慢,患者通常就诊延迟。KD在影像学上可能类似于其他慢性炎症性疾病,如结核病、血管畸形和肿瘤。临床相关性、组织学评估以及外周嗜酸性粒细胞增多和血清IgE水平升高被认为对确诊很重要。我们报告一例右侧下颌下区域无痛性肿胀并延伸至右侧腮腺浅叶的病例。血液学检查报告在正常范围内。超声(USG)、磁共振成像(MRI)和磁共振血管造影(MRA)倾向于诊断为静脉畸形。然而,切除病变的组织病理学检查确诊为KD。该病例证明即使在没有外周嗜酸性粒细胞增多和/或血清IgE水平升高的情况下,KD也有可能发生,并且在影像学检查中可能类似于静脉畸形。因此,即使在没有外周嗜酸性粒细胞增多和/或IgE水平升高的情况下,KD也必须列入无痛性颈部孤立性肿胀的鉴别诊断范围。

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本文引用的文献

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Kimura's disease: an unusual case of neck mass.木村病:一例罕见的颈部肿块病例。
Indian J Otolaryngol Head Neck Surg. 2008 Dec;60(4):353-5. doi: 10.1007/s12070-008-0101-2. Epub 2008 Nov 7.
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Kimura's disease.木村病
Indian J Otolaryngol Head Neck Surg. 2004 Jan;56(1):43-5. doi: 10.1007/BF02968773.

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