Yamamoto Tetsurou, Minamiguchi Sachiko, Watanabe Yoshiki, Tsuji Jun, Asato Ryo, Manabe Toshiaki, Haga Hironori
Department of Pathology, National Hospital Organization Kyoto Medical Center, 1-1 Mukaihata-cho Fukakusa Fushimi-ku, Kyoto-shi, Kyoto, 612-8555, Japan,
Head Neck Pathol. 2014 Jun;8(2):198-203. doi: 10.1007/s12105-013-0482-8. Epub 2013 Aug 2.
Kimura disease (KD) affecting an unusual site is a diagnostic challenge. We report herein the case of a 62-year-old Japanese woman who presented with swelling of the epiglottis, resulting in airway narrowing. Microscopically, biopsied and resected specimens both revealed lymphoid proliferation of a reactive immunophenotype, accompanied by vascular proliferation, eosinophilic infiltration, and stromal sclerosis. Adjunctive immunohistochemistry with immunoglobulin E in addition to laboratory and histological findings led us to seriously consider a diagnosis of KD. The patient underwent surgical removal with postoperative steroid therapy and has no evidence of recurrence. Our experience suggests that KD is potentially fatal as well as showing difficulty in the histological diagnosis when occurring in the upper respiratory tract, such as the epiglottis. A literature review disclosed that our case is the 11th case so far reported in this location, and that KD of the epiglottis did not show any male preponderance, as seen in other places.
木村病（KD）累及不寻常部位是一项诊断挑战。我们在此报告一例62岁日本女性病例，该患者表现为会厌肿胀，导致气道狭窄。显微镜下，活检和切除标本均显示具有反应性免疫表型的淋巴样增生，伴有血管增生、嗜酸性粒细胞浸润和间质硬化。除实验室检查和组织学结果外，免疫球蛋白E辅助免疫组化使我们高度怀疑KD的诊断。该患者接受了手术切除并术后接受类固醇治疗，目前无复发迹象。我们的经验表明，KD发生在上呼吸道（如会厌）时，不仅组织学诊断困难，还可能致命。文献回顾显示，我们的病例是迄今为止该部位报道的第11例，且会厌部的KD未表现出如其他部位那样的男性优势。
