Department of Gastroenterology, Centro Hospitalar Universitário do Porto, Porto, Portugal.
Unit of Clinical Immunology, Centro Hospitalar Universitário do Porto, Porto, Portugal.
J Gastrointestin Liver Dis. 2023 Jun 22;32(2):141. doi: 10.15403/jgld-4921.
A 67-year-old man with previous cardiovascular disease was referred to our consultation due to a 5-month history of recurrent epigastric pain. Esophagogastroduodenoscopy and full blood workup presented no alterations. CT scan showed an irregularly shaped mass at the root of the mesentery, measuring 40x25x47mm, with spiculated contours and retractile behaviour (a). Simultaneous densification of the adjacent fat and infracentimetric ganglionic formations scattered throughout the mesentery were shown. Surgical biopsy revealed extensive storiform fibrosclerosis, with the presence of interstitial lymphoplasmocytic infiltrate and obliterative phlebitis (b); the plasma cells had mostly IgG expression, with IgG4:IgG ratio >40% (c), accounting for more than 30- 40 IgG4 plasma cells per field. The serum IgG4 level was 137mg/dL. A diagnosis of IgG4-related sclerosing mesenteritis was made, without other organ involvement. Prednisolone (0.6mg/kg/d) improved partially the abdominal pain, so steroid sparing strategy with off-label rituximab was associated. Due to its low prevalence, the understanding of this entity is scarce, and its diagnosis is challenging. Unlike other manifestations of IgG4-related disease, the intra-abdominal disease is identified in later stages, due to unspecific symptoms. This case aims to raise awareness about this condition as a differential diagnosis of abdominal pain.
一位 67 岁的男性,有心血管疾病病史,因反复发作性上腹痛 5 个月而被转介至我处就诊。食管胃十二指肠镜检查和全血细胞检查均未见异常。CT 扫描显示肠系膜根部有一形状不规则的肿块,大小为 40x25x47mm,呈刺状轮廓,有回缩行为(a)。同时显示相邻脂肪和散布在肠系膜中的亚厘米级神经节形成物的密集化。外科活检显示广泛的束状纤维硬化,伴有间质淋巴浆细胞浸润和闭塞性静脉炎(b);浆细胞主要表达 IgG,IgG4:IgG 比值>40%(c),每个视野超过 30-40 个 IgG4 浆细胞。血清 IgG4 水平为 137mg/dL。诊断为 IgG4 相关硬化性肠系膜炎,无其他器官受累。泼尼松龙(0.6mg/kg/d)部分改善了腹痛,因此采用了类固醇保留策略,联合使用利妥昔单抗。由于其患病率低,对这种疾病的认识还很有限,其诊断具有挑战性。与 IgG4 相关疾病的其他表现不同,由于症状不特异,腹腔内疾病在晚期才被识别。本病例旨在提高对这种疾病的认识,作为腹痛的鉴别诊断。