Horiguchi Hidehisa, Nakata Satoshi, Nobusawa Sumihito, Uyama Shinichi, Miyamoto Tadashi, Ueta Hiromi, Fujimoto Naomi, Yokoo Hideaki
Department of Laboratory Medicine and Pathology, Tokushima Municipal Hospital, Tokushima, Japan.
Department of Human Pathology, Gunma University Graduate School of Medicine, Maebashi, Japan.
Neuropathology. 2017 Feb;37(1):52-57. doi: 10.1111/neup.12317. Epub 2016 Jun 14.
Atypical teratoid/rhabdoid tumors (AT/RTs) are rare malignant neoplasms of the CNS that preferentially affect young children. We herein report an adult case of AT/RT surviving for more than 5 years with the residual tumor. The patient, a 24-year-old man at onset, presented with a contrast-enhancing mass lesion in the left occipital lobe, and underwent partial tumor resection. Histologically, the tumor was predominantly composed of long spindle cells exhibiting nuclear palisading and perivascular pseudorosettes, which appeared to mimic mesenchymal, ependymal and Schwann cell tumors. A small number of isolated rhabdoid cells did not initially attract attention, and a tentative pathological diagnosis of a malignant mesenchymal tumor was made. In a later examination focusing on the small areas of rhabdoid cells, the extensive loss of the nuclear expression of INI1 was detected in all areas. Diffuse staining with vimentin and focal immunoreactivity for epithelial membrane antigen and alpha smooth muscle actin were observed not only in AT/RT foci, but also in spindle cell areas. Thus, polyphenotypic immunoreactivity was evident. Fluorescence in situ hybridization revealed a homozygous deletion of chromosome 22q covering the INI1 locus. Histopathological differences between infant and adult AT/RTs currently remain unclear. In the case of a malignant adult brain tumor showing a hardly classifiable morphology and immunophenotypic diversity, an analysis of the INI1 status may contribute to an accurate diagnosis.
非典型畸胎样/横纹肌样肿瘤(AT/RTs)是中枢神经系统罕见的恶性肿瘤,好发于幼儿。我们在此报告一例成年AT/RT患者,带瘤生存超过5年。患者发病时为24岁男性,表现为左枕叶强化肿块,接受了部分肿瘤切除术。组织学上,肿瘤主要由长梭形细胞组成,可见核栅栏状排列和血管周围假菊形团,这似乎类似于间叶性、室管膜性和施万细胞肿瘤。少数孤立的横纹肌样细胞最初未引起注意,初步病理诊断为恶性间叶性肿瘤。在后来针对横纹肌样细胞小区域的检查中,所有区域均检测到INI1核表达广泛缺失。波形蛋白弥漫性染色以及上皮膜抗原和α平滑肌肌动蛋白的局灶性免疫反应不仅在AT/RT灶中观察到,在梭形细胞区域也有观察到。因此,多表型免疫反应明显。荧光原位杂交显示覆盖INI1位点的22q染色体纯合缺失。目前婴儿和成人AT/RTs的组织病理学差异仍不清楚。对于形态难以分类且免疫表型多样的成年恶性脑肿瘤病例,INI1状态分析可能有助于准确诊断。