Faculty of Medical Sciences of Minas Gerais, Alameda Ezequiel Dias, 275, Belo Horizonte, MG, Brazil.
Department of Oral Surgery and Pathology, School of Dentistry, Universidade Federal de Minas Gerais, Avenida Presidente Antônio Carlos, Belo Horizonte, MG, 6627, Brazil.
Head Neck Pathol. 2024 Sep 30;18(1):90. doi: 10.1007/s12105-024-01696-5.
Striated duct adenoma (SDA) is a rare benign salivary gland tumor with a recently described genetic signature. Recurrent oncogenic mutations affecting the IDH2 gene differentiate SDA from its primary differential diagnosis of canalicular adenoma. Here, we report a case of SDA affecting the parotid gland with IDH1/2 mutation-specific immunohistochemical positivity. Additionally, we provide a scoping review developed according to the Cochrane Methodology and reported following the Joana Briggs Institute (JBI) checklist to synthesize all previously published cases of SDA. The review protocol was registered on the Open Science Framework (OSF) platform ( https://osf.io/7mztg ). The searches were performed using Medline, Embase, Web of Science, and LILACS, with no date or language limit. Studies were evaluated for eligibility, extracted, and compiled in a narrative form. Seven studies with 20 patients with SDA, including ours, were analyzed. The tumors mainly affected the parotid gland (13/20) in patients with a mean age of 62 years and did not display sex predilection. Swelling was the leading clinical symptom. The mean follow-up duration was 26 months with no recurrence or metastasis after resection.
Awareness of the clinicopathological features and the use of IDH1/2 mutation-specific immunohistochemistry are pivotal for the consistent identification of SDA, and assessment for true biological potential will require increased follow-up and scrutiny.
纹管性腺瘤(SDA)是一种罕见的良性涎腺肿瘤,具有最近描述的遗传特征。影响 IDH2 基因的复发性致癌突变将 SDA 与其主要鉴别诊断——管腔性腺瘤区分开来。在这里,我们报告了一例发生于腮腺的 SDA,具有 IDH1/2 突变特异性免疫组织化学阳性。此外,我们根据 Cochrane 方法学和 Joana Briggs 研究所(JBI)清单提供了一份范围综述,以综合所有以前发表的 SDA 病例。该综述方案已在开放科学框架(OSF)平台(https://osf.io/7mztg)上注册。使用 Medline、Embase、Web of Science 和 LILACS 进行了检索,没有日期或语言限制。对符合条件的研究进行了评估、提取,并以叙述形式进行了汇编。分析了包括我们的研究在内的 7 项研究,共 20 例 SDA 患者。肿瘤主要发生在腮腺(13/20),患者平均年龄为 62 岁,无性别偏好。肿胀是主要的临床症状。平均随访时间为 26 个月,切除后无复发或转移。
了解临床病理特征和使用 IDH1/2 突变特异性免疫组织化学是一致识别 SDA 的关键,评估其真正的生物学潜能需要增加随访和审查。