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1型神经纤维瘤病患者的软组织束膜瘤及其他罕见肿瘤

Soft tissue perineurioma and other unusual tumors in a patient with neurofibromatosis type 1.

作者信息

Schaefer Inga-Marie, Ströbel Philipp, Thiha Aung, Sohns Jan Martin, Mühlfeld Christian, Küffer Stefan, Felmerer Gunther, Stepniewski Adam, Pauli Silke, Agaimy Abbas

机构信息

Institute of Pathology, University Medical Center Göttingen Germany ; Department of Pathology, Brigham and Women's Hospital, Harvard Medical School Boston MA, USA.

出版信息

Int J Clin Exp Pathol. 2013 Nov 15;6(12):3003-8. eCollection 2013.

Abstract

Perineurioma is a rare benign peripheral nerve sheath tumor featuring perineurial differentiation. Perineurioma occurs sporadically with only one reported case in the setting of neurofibromatosis type 1 (NF-1). We present a 6.7-cm soft tissue perineurioma of the lower leg in a 51-year-old man with proven NF-1. The tumor displayed whorled and fascicular pattern with infiltrative margins and expressed EMA, GLUT-1, claudin-1, and CD34. Electron microscopy confirmed diagnosis. Furthermore, lipomatosis, cutaneous angiomatous nodules, vasculopathy, and iliac spine lesion consistent with non-ossifying fibroma were observed. Tumor DNA revealed no NF2 mutations or chromosomal aberrations but a germline NF1-deletion (c.449_502delTGTT) was detected in his blood sample. His brother displayed neurofibromas, duodenal ganglioneuroma and colonic juvenile polyp, and his mother a neurofibroma, cutaneous squamous cell carcinoma, and jejunal gastrointestinal stromal tumor (GIST); both were affected by NF-1. In conclusion, perineurioma may rarely be NF-1 related and should be included in the spectrum of neoplasms occurring in this disorder.

摘要

神经束膜瘤是一种罕见的良性周围神经鞘瘤,具有神经束膜分化特征。神经束膜瘤散发性发生,仅1例报道发生于1型神经纤维瘤病(NF-1)患者。我们报告1例51岁确诊为NF-1的男性患者,其小腿出现一个6.7 cm的软组织神经束膜瘤。肿瘤呈漩涡状和束状结构,边界浸润性生长,表达上皮膜抗原(EMA)、葡萄糖转运蛋白1(GLUT-1)、闭合蛋白-1(claudin-1)和CD34。电镜检查确诊。此外,还观察到脂肪瘤病、皮肤血管瘤性结节、血管病变以及与非骨化性纤维瘤一致的髂骨病变。肿瘤DNA检测未发现NF2突变或染色体畸变,但在其血液样本中检测到一个种系NF1缺失(c.449_502delTGTT)。他的哥哥患有神经纤维瘤、十二指肠神经节瘤和结肠幼年性息肉,他的母亲患有神经纤维瘤、皮肤鳞状细胞癌和空肠胃肠道间质瘤(GIST);两人均患有NF-1。总之,神经束膜瘤可能很少与NF-1相关,应纳入该疾病相关肿瘤谱。

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