Hirokawa Mitsuyoshi, Takada Nami, Abe Hideyuki, Suzuki Ayana, Higuchi Miyoko, Miya Akihiro, Hayashi Toshitetsu, Fukushima Mitsuhiro, Kawahara Akihiko, Miyauchi Akira
Department of Diagnostic Pathology and Cytology, Kuma Hospital, 8-2-35 Shimoyamate-dori, Chuo-Ku, Kobe, Hyogo 650-0011, Japan.
Department of Laboratory Medicine, Kuma Hospital, 8-2-35 Shimoyamate-dori, Chuo-Ku, Kobe, Hyogo 650-0011, Japan.
Endocr J. 2018 Apr 26;65(4):427-436. doi: 10.1507/endocrj.EJ17-0462. Epub 2018 Feb 8.
We report three cases of thyroid sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE), which is an extremely rare variant of mucoepidermoid carcinoma (MEC). The aims of this report were to describe the clinicopathological findings, including results from immunohistochemical and fluorescence in situ hybridization analysis of thyroid SMECE, as well as to discuss the distinction between thyroid SMECE and its salivary counterpart. The cases included a 63-year-old female, a 44-year-old male, and a 66-year-old female, with all patients presenting with Hashimoto's thyroiditis. Nodal metastasis was not found in any of the three cases. Neither regional recurrences nor distant metastases were found in any patient during the follow-up, which was 20 years, 3 years, and 18 months, respectively. Histologically, tumors were composed of epidermoid carcinoma cells, intermediate type carcinoma cells, and goblet cell-type mucus-secreting carcinoma cells, with all tumors displaying a sclerotic stroma with eosinophilic and lymphocytic infiltration. The formation of eosinophilic abscess in the tumor nests that might be a novel characteristic finding of SMECE was observed. Immunohistochemically, the carcinoma cells were positive for cytokeratin 34βE12, TTF-1, and PAX8, but negative for thyroglobulin. In two cases, increased IgG4-positive plasma cells were observed. Mastermind-like transcriptional coactivator 2 (MAML2), according to fluorescence in situ hybridization, was intact in all cases. In conclusion, thyroid SMECE has favorable outcomes and seems to be genetically different from salivary MEC. This is the first report to describe the presence of increased IgG4-positive plasma cells in the stroma of SMECE.
我们报告了3例伴有嗜酸性粒细胞增多的甲状腺硬化性黏液表皮样癌(SMECE),这是黏液表皮样癌(MEC)的一种极其罕见的变异型。本报告的目的是描述临床病理特征,包括甲状腺SMECE的免疫组织化学和荧光原位杂交分析结果,并讨论甲状腺SMECE与其涎腺对应物之间的区别。病例包括一名63岁女性、一名44岁男性和一名66岁女性,所有患者均患有桥本甲状腺炎。3例均未发现淋巴结转移。在分别为20年、3年和18个月的随访期间,所有患者均未发现局部复发或远处转移。组织学上,肿瘤由表皮样癌细胞、中间型癌细胞和杯状细胞型黏液分泌癌细胞组成,所有肿瘤均显示有硬化性间质,并伴有嗜酸性粒细胞和淋巴细胞浸润。观察到肿瘤巢内形成嗜酸性脓肿,这可能是SMECE的一个新的特征性表现。免疫组织化学检查显示,癌细胞细胞角蛋白34βE12、甲状腺转录因子-1(TTF-1)和配对盒基因8(PAX8)呈阳性,但甲状腺球蛋白呈阴性。2例观察到IgG4阳性浆细胞增多。荧光原位杂交显示,所有病例中类主脑样转录共激活因子2(MAML2)均完整。总之,甲状腺SMECE预后良好,似乎在基因上与涎腺MEC不同。这是第一篇描述SMECE间质中IgG4阳性浆细胞增多的报告。