Department of Endocrinology, Hebei General Hospital, Shijiazhaung, China.
Department of Endocrinology, Hebei General Hospital, Shijiazhaung, China
Ann Clin Lab Sci. 2020 Nov;50(6):825-833.
The present study reported a rare case with thymic carcinoid as the first manifestation of multiple endocrine neoplasia type 1 (MEN1) syndrome, which presented with gene mutations of the MEN1 and glucokinase regulatory protein (GCKR). In this report, a 40-year-old male was diagnosed as MEN1 syndrome with thymic carcinoid, pancreatic cancer, hyperparathyroidism, and insulinoma with intrahepatic metastasis. Genetic testing showed the mutations of the MEN1 (c.378 G>A, p. Trp126*) in the patient, his children and two sisters, and GCKR (c.151C>T, p. Arg51*) gene in the patient and his children. The pathological examination showed that neuroendocrine tumor (NET) of the pancreas was characterized as 6 mitoses per 10 high-power fields (HPF), infiltration of adipose tissue, no intravascular tumor thrombus and nerve infiltration. In addition, NET of the liver was characterized as 4 mitoses per 10 HPF, no intravascular tumor thrombus and nerve infiltration. Immunohistochemical staining showed Ckpan (+), Syn (+), CgA (+), CD 56 (+), PGP 9.5 (+), and Ki67-positive cells (8-10%) in NET. Therefore, we suggested that genetic testing in family members of MEN1 patient, which may be helpful for early diagnosis.
本研究报道了一例罕见病例,以胸腺癌为多发性内分泌肿瘤 1 型(MEN1)综合征的首发表现,该患者同时存在 MEN1 和葡萄糖激酶调节蛋白(GCKR)基因突变。本报告中,一名 40 岁男性被诊断为 MEN1 综合征,合并胸腺癌、胰腺癌、甲状旁腺功能亢进症和肝内转移的胰岛素瘤。基因检测显示患者及其子女和两位姐妹均存在 MEN1(c.378 G>A,p.Trp126*)突变,患者及其子女存在 GCKR(c.151C>T,p.Arg51*)基因突变。病理检查显示胰腺神经内分泌肿瘤(NET)具有 6 个/10 高倍视野(HPF)的有丝分裂、脂肪组织浸润、无血管内肿瘤血栓和神经浸润。此外,肝 NET 具有 4 个/10 HPF 的有丝分裂、无血管内肿瘤血栓和神经浸润。免疫组织化学染色显示 NET 中 Ckpan(+)、Syn(+)、CgA(+)、CD56(+)、PGP9.5(+)和 Ki67 阳性细胞(8-10%)。因此,我们建议对 MEN1 患者的家族成员进行基因检测,这可能有助于早期诊断。