Ito Hiroyuki, Mishima Yusuke, Cho Tsubomi, Ogiwara Naoki, Shinma Yoshimasa, Yokota Masashi, Anzai Kazuya, Tsuda Shingo, Nagata Junko, Kojima Seiichiro, Sasaki Noriko, Wakabayashi Takayuki, Watanabe Norihito, Suzuki Takayoshi
Department of Gastroenterology, Tokai University Hachioji Hospital, Tokyo, Japan.
Department of Rheumatology, Tokai University Hachioji Hospital, Tokyo, Japan.
Case Rep Gastroenterol. 2020 Dec 11;14(3):668-674. doi: 10.1159/000511863. eCollection 2020 Sep-Dec.
We report a case of eosinophilic cholecystitis associated with eosinophilic granulomatosis with polyangiitis (EGPA) complicated by cerebral hemorrhage. A 60-year-old man presented to a local hospital with a diagnosis of acute cholecystitis, with persistent fever and epigastric pain for 2 weeks. His symptoms persisted despite 3-week hospitalization; therefore, he was transferred to our hospital for further evaluation. Laboratory investigations upon admission showed white blood cells 26,300/µL and significant eosinophilia (eosinophils 61%). Abdominal computed tomography revealed no gallbladder enlargement but a circumferentially edematous gallbladder wall. Additional blood test results were negative for antineutrophil cytoplasmic and perinuclear antineutrophil cytoplasmic antibodies; however, immunoglobulin (Ig)G and IgE levels were high at 1,953 mg/dL and 3,040/IU/mL, respectively. He improved following endoscopic transnasal gallbladder drainage for cholecystitis and was diagnosed with EGPA and received corticosteroid and immunosuppressant combination therapy. The eosinophil count decreased immediately after treatment, and abdominal pain and numbness resolved. He returned with left-sided suboccipital hemorrhage likely attributed to EGPA 6 months after discharge. EGPA is characterized by inflammation of small blood vessels and clinically manifests with an allergic presentation of bronchial asthma, as well as renal dysfunction, interstitial pneumonia, enteritis, and cerebral hemorrhage. Few reports have described cholecystitis as a presenting symptom of EGPA. We report a rare case of such a presentation with added considerations.
我们报告一例与嗜酸性肉芽肿性多血管炎(EGPA)相关的嗜酸性胆囊炎,并伴有脑出血。一名60岁男性因急性胆囊炎就诊于当地医院,持续发热和上腹部疼痛2周。尽管住院3周,其症状仍持续存在;因此,他被转至我院作进一步评估。入院时实验室检查显示白细胞计数为26,300/µL,且有明显的嗜酸性粒细胞增多(嗜酸性粒细胞占61%)。腹部计算机断层扫描显示胆囊无增大,但胆囊壁呈环形水肿。抗中性粒细胞胞浆抗体和核周抗中性粒细胞胞浆抗体的其他血液检测结果均为阴性;然而,免疫球蛋白(Ig)G和IgE水平分别高达1,953 mg/dL和3,040/IU/mL。他接受了内镜经鼻胆囊引流治疗胆囊炎后病情好转,并被诊断为EGPA,接受了皮质类固醇和免疫抑制剂联合治疗。治疗后嗜酸性粒细胞计数立即下降,腹痛和麻木症状消失。出院6个月后,他因可能归因于EGPA的左侧枕下出血再次就诊。EGPA的特征是小血管炎症,临床表现为支气管哮喘的过敏表现,以及肾功能不全、间质性肺炎、肠炎和脑出血。很少有报告将胆囊炎描述为EGPA的首发症状。我们报告了这样一例罕见的病例,并增加了一些考虑因素。