Sakaguchi N, Sano K, Ito M, Baba T, Fukuzawa M, Hotchi M
Department of Pathology, Shinshu University School of Medicine, Matsumoto, Japan.
Am J Surg Pathol. 1996 Jul;20(7):889-97. doi: 10.1097/00000478-199607000-00013.
A 48-year-old man with neurofibromatosis type 1 presented with chest pain, paroxysmal hypertension, tachycardia, and progressive respiratory insufficiency. Clinical investigation displayed calcified tumors in the anterior mediastinum and pararenal region. Histological examination at autopsy revealed composite tumors consisting of pheochromocytoma and malignant peripheral nerve sheath tumor (MPNST) at two sites: the left adrenal gland and the region surrounding the inferior vena cava, probably corresponding to the right adrenal gland. The MPNST component showed a varied histological appearance, including hyalinized bands with polygonal cells, a cartilaginous and myxoid stroma, a hemangiopericytomatous architecture, and a fibrosarcomatous structure, which suggested osteosarcoma, chondrosarcoma, angiosarcoma, and fibrosarcoma, respectively. In addition, based on the ultrastructural findings, the gastrointestinal tract was involved with mesenchymal tumors showing neurogenic differentiation. These lesions suggest the divergent cellular differentiation of neural crest-derived cells to mesenchymal elements as well as neuroectodermal neoplasms.
一名48岁的1型神经纤维瘤病男性患者,出现胸痛、阵发性高血压、心动过速和进行性呼吸功能不全。临床检查显示前纵隔和肾周区域有钙化肿瘤。尸检组织学检查发现两个部位存在由嗜铬细胞瘤和恶性外周神经鞘瘤(MPNST)组成的复合肿瘤,分别位于左肾上腺和下腔静脉周围区域,可能对应右肾上腺。MPNST成分呈现出多样的组织学表现,包括带有多边形细胞的玻璃样化带、软骨样和黏液样基质、血管外皮细胞瘤样结构以及纤维肉瘤样结构,分别提示骨肉瘤、软骨肉瘤、血管肉瘤和纤维肉瘤。此外,基于超微结构发现,胃肠道存在显示神经源性分化的间叶性肿瘤。这些病变提示神经嵴衍生细胞向间叶成分以及神经外胚层肿瘤的不同细胞分化。