Department of Oral and Maxillofacial Surgery, Peking University School and Hospital of Stomatology, National Engineering Laboratory for Digital and Material Technology of Stomatology, Beijing Key Laboratory of Digital Stomatology, Beijing, China.
Department of Oral Radiology, Peking University School and Hospital of Stomatology, National Engineering Laboratory for Digital and Material Technology of Stomatology, Beijing Key Laboratory of Digital Stomatology, Beijing, China.
Int J Oral Maxillofac Surg. 2021 Jul;50(7):895-905. doi: 10.1016/j.ijom.2020.05.023. Epub 2020 Dec 29.
The aim of this study was to investigate key points for the differential diagnosis of immunoglobulin G4-related sialadenitis (IgG4-RS) and Kimura's disease (KD) involving the salivary glands. The clinical, serological, radiological, histological, and immunohistochemical features of 85 IgG4-RS cases and 52 KD cases were evaluated comparatively. Seventy-two IgG4-RS cases had enlargement of multiple salivary and/or lacrimal glands; 67 patients had bilateral submandibular gland (SMG) involvement. Unilateral parotid gland involvement (59.6%) and comorbid skin lesions (61.5%) were common in KD. Serum IgG4 was elevated in 94.1% of IgG4-RS cases versus 19.0% of KD cases (cut-off value=266.5mg/dl). KD was more commonly associated with elevated eosinophil counts (86% vs 23.1%) and elevated IgE concentrations (95.5% vs 76.6%). Storiform fibrosis, irregular lymphoid follicles, and increased IgG4-positive cells (112.9±37.6/high-power field (HPF)) were common in IgG4-RS. Acellular fibrosis, regular lymphoid follicles, IgE-positive reticular networks, increased IgE-positive cells (43.4±26.7/HPF), and tryptase-positive mast cells (29.7±13.3/HPF) were usually detected in KD. Computed tomography showed that 85.7% of KD cases involved subcutaneous fat tissue. A superficial hypoechoic and reticular pattern with multiple hypoechoic foci were the sonographic features of the SMG in IgG4-RS. Despite numerous overlapping manifestations, histopathological examination showed meaningful differences in the types of fibrosis, eosinophils, and IgG4-positive cell counts. Comprehensive evaluation of clinical, serological, radiological, and histopathological features are crucial for the differential diagnosis.
本研究旨在探讨免疫球蛋白 G4 相关唾液腺炎（IgG4-RS）和 Kimura 病（KD）累及唾液腺的鉴别诊断要点。比较了 85 例 IgG4-RS 病例和 52 例 KD 病例的临床、血清学、影像学、组织学和免疫组织化学特征。72 例 IgG4-RS 病例有多个唾液腺和/或泪腺肿大；67 例患者双侧颌下腺（SMG）受累。单侧腮腺受累（59.6%）和伴发皮肤病变（61.5%）是 KD 的常见特征。血清 IgG4 在 94.1%的 IgG4-RS 病例中升高，而在 19.0%的 KD 病例中升高（临界值=266.5mg/dl）。KD 更常伴有嗜酸性粒细胞计数升高（86% vs 23.1%）和 IgE 浓度升高（95.5% vs 76.6%）。IgG4 阳性细胞（112.9±37.6/高倍视野（HPF））增多的席纹状纤维化、不规则淋巴滤泡和增多的 IgG4 阳性细胞是 IgG4-RS 的常见特征。无细胞纤维化、规则的淋巴滤泡、IgE 阳性网状网络、增多的 IgE 阳性细胞（43.4±26.7/HPF）和 tryptase 阳性肥大细胞（29.7±13.3/HPF）通常在 KD 中检测到。计算机断层扫描显示 85.7%的 KD 病例累及皮下脂肪组织。SMG 的超声特征是皮下组织呈低回声和网状，伴有多个低回声灶。尽管存在许多重叠表现，但组织病理学检查显示纤维化、嗜酸性粒细胞和 IgG4 阳性细胞计数的类型存在有意义的差异。全面评估临床、血清学、影像学和组织病理学特征对于鉴别诊断至关重要。
