Division of Rheumatology, Department of Internal Medicine, Faculty of Medicine, Eskişehir Osmangazi University, Eskişehir, 26480, Turkey.
Division of Endocrinology, Department of Internal Medicine, Faculty of Medicine, Kütahya Health Science University, Kütahya, Turkey.
Rheumatol Int. 2022 Jul;42(7):1297-1305. doi: 10.1007/s00296-021-05076-y. Epub 2022 Jan 7.
Immunoglobulin (Ig) G4-related disease (IgG4RD) is a chronic autoimmune disorder characterized by dense lymphoplasmacytic infiltrations and fibrosis of storiform pattern. The most typical manifestations include major salivary or lacrimal gland involvement, autoimmune pancreatitis, and retroperitoneal fibrosis. While the increase in IgG4 is the typical feature of the disease, hypercalcemia has been rarely reported in IgG4RD so far, only one of these cases has been shown parathyroid gland involvement (isolated involvement). In this study, we present a 43-year-old female patient with weight loss, pancreatic mass, lymphadenopathy, nodular lesion in the lung, hypercalcemia, and also increased level of serum IgG4. Histopathological investigation following parathyroidectomy revealed a dense lymphoplasmacytic infiltrate with an IgG4 to IgG ratio of > 50% in the fat tissue surrounding the parathyroid gland, particularly at the perivascular areas. This is the first systemic IgG4RD case in combination with hypercalcemia in the literature who was detected to have parathyroid adenoma. Our aim in this review is to emphasize that, although rarely, IgG4RD may be accompanied by hypercalcemia and parathyroid gland may be one of its target sites.
免疫球蛋白（Ig）G4 相关疾病（IgG4RD）是一种慢性自身免疫性疾病，其特征为致密的淋巴浆细胞浸润和席纹状纤维化。最典型的表现包括大涎腺或泪腺受累、自身免疫性胰腺炎和腹膜后纤维化。虽然 IgG4 的增加是该疾病的典型特征，但迄今为止 IgG4RD 中的高钙血症很少见，仅有一例显示甲状旁腺受累（孤立性受累）。在本研究中，我们报告了一例 43 岁女性患者，其表现为体重减轻、胰腺肿块、淋巴结病、肺部结节性病变、高钙血症和血清 IgG4 水平升高。甲状旁腺切除术后的组织病理学检查显示，甲状旁腺周围脂肪组织中存在致密的淋巴浆细胞浸润，IgG4 与 IgG 的比值>50%，特别是在血管周围区域。这是文献中首例系统性 IgG4RD 合并高钙血症并检测到甲状旁腺腺瘤的病例。我们在本次综述中的目的是强调，尽管很少见，但 IgG4RD 可能伴有高钙血症，甲状旁腺可能是其靶器官之一。
