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木村病先后累及头颈部多个部位:一例随访13年的病例报告及文献复习

Kimura's disease sequentially involving multiple sites in the head and neck: A case report with a 13-year follow-up and literature review.

作者信息

Li Rui-Si, Zhang Xiang-Zi, Quan Yu-Hua, Xuan Yun-Ze

机构信息

Department of Dentistry, Yanbian University Hospital, China.

Oral Medicine Department, Yanbian University College, China.

出版信息

J Int Med Res. 2025 May;53(5):3000605251337422. doi: 10.1177/03000605251337422. Epub 2025 May 13.

Abstract

Kimura's disease is a rare, chronic inflammatory condition with a high recurrence rate, primarily affecting young to middle-aged Asian males. It typically manifests as masses in the head and neck, accompanied with regional lymphadenopathy. This report describes the case of a 50-year-old man initially diagnosed with Kimura's disease 13 years ago, following surgical removal of a left submandibular mass. Subsequent recurrences involved the parotid and contralateral submandibular regions, requiring radiotherapy and intermittent oral prednisone therapy. After 6 years, he developed progressive proptosis and visual impairment, revealing multiple orbital masses bilaterally. A puncture examination of the left submandibular mass was indicative of Kimura's disease. Throughout the follow-up period, the eosinophil levels correlated with prednisone use. This case highlights the disease's capacity for multisite recurrence within the head and neck over an extended duration. The extensive orbital involvement without renal manifestations is a rare presentation. Long-term follow-up is crucial in Kimura's disease management, and oral prednisone can effectively control disease progression.

摘要

木村病是一种罕见的慢性炎症性疾病,复发率高,主要影响年轻至中年的亚洲男性。它通常表现为头颈部肿块,并伴有局部淋巴结病。本报告描述了一名50岁男性的病例,该患者13年前最初被诊断为木村病,此前手术切除了左侧下颌下肿块。随后的复发累及腮腺和对侧下颌下区域,需要放疗和间歇性口服泼尼松治疗。6年后,他出现进行性眼球突出和视力损害,双侧发现多个眼眶肿块。左侧下颌下肿块的穿刺检查提示为木村病。在整个随访期间,嗜酸性粒细胞水平与泼尼松的使用相关。该病例突出了该疾病在头颈部长期多部位复发的能力。广泛的眼眶受累而无肾脏表现是一种罕见的表现。长期随访对木村病的管理至关重要,口服泼尼松可有效控制疾病进展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/5de7/12075989/5d2cfc5ee4fe/10.1177_03000605251337422-fig1.jpg

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