Li Chun-Yu, Han Ya-Mei, Xu Ke, Wu Shu-Yue, Lin Xu-Yong, Cao Hong-Yi
Department of Pathology, The First Affiliated Hospital and College of Basic Medical Sciences, China Medical University, Shenyang, 110001, People's Republic of China.
Onco Targets Ther. 2021 Mar 29;14:2227-2233. doi: 10.2147/OTT.S302613. eCollection 2021.
In adults, yolk sac tumors (YSTs) in the nasal cavity and paranasal sinuses are very rare. To date, only six cases have been reported in the English literature. YSTs in adults are often accompanied by cancer, teratocarcinosarcoma, and other malignant components. Here, we have reported a case of nasal tumor in a 55-year-old man with nasal obstruction and epistaxis. Morphologically, the tumor showed histological characteristics of pure YST. Immunohistochemical staining showed diffuse expression of SALL4, CDX2, and GPC-3 accompanied by sporadic expression of alpha-fetoprotein (AFP) and CD117. After 20 and 40 days of operation, the serum AFP level was 220.30 and 43.60 ng/mL (normal, <7 ng/mL), respectively, which supported the pathological diagnosis of YST. However, we further performed immunohistochemical staining and fluorescence in situ hybridization using an INI-1 probe to detect the status of INI-1 in tumor cells. The results revealed that INI-1 was absent in tumor cells. Hence, we corrected the diagnosis to SMARCB1 (INI-1)-deficient carcinoma of the nasal cavity with YST differentiation. The patient underwent surgery and adjuvant radiotherapy in our hospital without evidence of recurrence or metastasis at the 6-month follow-up. The serum AFP level had also normalized. In conclusion, our case demonstrates that INI-1-deficient carcinoma may exhibit, a pure YST differentiation and immunophenotype, and elevated serum AFP levels. In adults, YST in the nasal cavity may represent INI-1-deficient carcinoma, which may be a potential diagnostic pitfall.
在成人中,鼻腔和鼻窦的卵黄囊瘤(YSTs)非常罕见。迄今为止,英文文献中仅报道了6例。成人YSTs常伴有癌、畸胎癌肉瘤和其他恶性成分。在此,我们报告了1例55岁男性鼻腔肿瘤患者,该患者有鼻塞和鼻出血症状。形态学上,肿瘤表现为纯YST的组织学特征。免疫组化染色显示SALL4、CDX2和GPC-3弥漫性表达,同时伴有甲胎蛋白(AFP)和CD117散在表达。术后20天和40天,血清AFP水平分别为220.30和43.60 ng/mL(正常,<7 ng/mL),这支持了YST的病理诊断。然而,我们进一步进行了免疫组化染色,并使用INI-1探针进行荧光原位杂交,以检测肿瘤细胞中INI-1的状态。结果显示肿瘤细胞中INI-1缺失。因此,我们将诊断修正为鼻腔SMARCB1(INI-1)缺陷型癌伴YST分化。该患者在我院接受了手术及辅助放疗,6个月随访时无复发或转移迹象。血清AFP水平也已恢复正常。总之,我们的病例表明,INI-1缺陷型癌可能表现出纯YST分化和免疫表型,以及血清AFP水平升高。在成人中,鼻腔YST可能代表INI-1缺陷型癌,这可能是一个潜在的诊断陷阱。