Kuwahara Toshiaki, Kobayashi Yoshiki, Yun Yasutaka, Kanda Akira, Asako Mikiya, Ueki Shigeharu, Iwai Hiroshi
Department of Otolaryngology, Kansai Medical University, Osaka, Japan.
Allergy Center, Kansai Medical University Hospital, Hirakata, Japan.
Allergy Rhinol (Providence). 2019 Aug 19;10:2152656719869607. doi: 10.1177/2152656719869607. eCollection 2019 Jan-Dec.
Eosinophilic cholecystitis (EC) is a rare condition that presents in a manner comparable to acute cholecystitis. The diagnosis is based on classical symptoms of cholecystitis with excessive eosinophilic infiltration within the gallbladder. EC has been reported alone or in combination with manifestations, such as eosinophilic gastrointestinal tract inflammation. However, association with airway inflammation in patients with EC is rare. We report the case of a 65-year-old man who had refractory eosinophilic chronic rhinosinusitis with bronchial asthma. A second endoscopic sinus surgery (ESS) was performed as treatment for recurrent nasal polyps. EC occurred while inhaled corticosteroids were reduced after ESS. Pathologic examination of the excised gallbladder demonstrated submucosal infiltration with a number of eosinophils. Furthermore, immunohistostaining revealed many galectin-10-positive cells in both the gallbladder mucosa and the paranasal sinus mucosa. Galectin-10 is a major constituent of human eosinophils, also known as the Charcot-Leyden crystal protein, which has been linked with eosinophilic inflammation. Interestingly, nasal polyps were reduced without any additional treatments 1 month after the cholecystectomy.
We experienced a rare case wherein EC onset occurred in a patient with refractory eosinophilic airway inflammation during inhaled corticosteroid tapering. Galectin-10 might help diagnose rare cases of eosinophilic inflammation in multiple organs.
嗜酸性粒细胞性胆囊炎(EC)是一种罕见疾病,其表现方式与急性胆囊炎相似。诊断基于胆囊炎的典型症状以及胆囊内嗜酸性粒细胞的过度浸润。EC 已被单独报道,或与嗜酸性粒细胞性胃肠道炎症等表现合并报道。然而,EC 患者与气道炎症相关的情况较为罕见。我们报告一例 65 岁男性患者,其患有难治性嗜酸性粒细胞性慢性鼻-鼻窦炎伴支气管哮喘。作为复发性鼻息肉的治疗方法,进行了第二次鼻内镜鼻窦手术(ESS)。在 ESS 后吸入性糖皮质激素减量时发生了 EC。切除胆囊的病理检查显示黏膜下有大量嗜酸性粒细胞浸润。此外,免疫组化染色显示胆囊黏膜和鼻旁窦黏膜中均有许多半乳糖凝集素-10 阳性细胞。半乳糖凝集素-10 是人类嗜酸性粒细胞的主要成分,也被称为夏科-莱登结晶蛋白,它与嗜酸性粒细胞炎症有关。有趣的是,胆囊切除术后 1 个月,鼻息肉在未进行任何额外治疗的情况下缩小了。
我们遇到了一例罕见病例,即在吸入性糖皮质激素减量期间,难治性嗜酸性粒细胞性气道炎症患者发生了 EC。半乳糖凝集素-10 可能有助于诊断多器官嗜酸性粒细胞炎症的罕见病例。