Chang S M, Ho W L
Department of Pathology, Taichung Veterans General Hospital, Taiwan, R.O.C.
Zhonghua Yi Xue Za Zhi (Taipei). 1994 Aug;54(2):122-30.
Malignant peripheral nerve sheath tumor (MPNST) is an aggressive tumor of neural origin. To improve our experience with this rare tumor, we reviewed 21 patients seen during a 12-year period at the pathological department of Taichung Veterans General Hospital.
Clinical data including age, sex, presence or absence of von Recklinghausen's disease, tumor location and size, presenting symptoms, surgical management and surveillance were summarized. Microscopically, we examined necrosis, mitosis, differentiation, the resection margin, and the presence or absence of neurofibromatous component.
There were 15 males and 6 females with age range of 9-86 years. Seven cases were associated with neurofibromatosis and 14 cases were solitary MPNST: Patients with coexistent neurofibromatosis were younger (mean age at diagnosis, 32 years) than patients with solitary MPNST (mean, 60.2 years). Tumors of the former group tended to be centrally located. Anaplastic alteration was found in the recurrent tumors of two patients both of whom died of the disease. Of the 21 cases, epithelioid differentiation and osteoid metaplasia were seen in two tumors respectively by optical microscopy. Patients with von Recklinghausen's disease and large incompletely resected tumors had the most dismal prognosis. Of five patients with neurofibromatosis who were observed for more than two years, only one survived and no recurrence was found. The remaining four patients died within 3-9 months. However, of 14 cases of solitary MPNST, only two patients died of disease and ten of them had survived beyond two years.
Our work revealed that patients with von Recklinghausen's disease, a large unresectable centrally located tumor with diffuse anaplastic alteration, had frequent recurrences and poor prognosis. Aggressive surgical procedure with free resection margin is suggested. Malignant transformation should be highly suspected when a painful, rapidly growing tumor developed.
恶性外周神经鞘膜瘤(MPNST)是一种起源于神经的侵袭性肿瘤。为了提高我们对这种罕见肿瘤的治疗经验,我们回顾了台中荣民总医院病理科在12年期间收治的21例患者。
总结临床资料,包括年龄、性别、是否患有冯雷克林霍增氏病、肿瘤位置和大小、首发症状、手术治疗及随访情况。显微镜下,我们检查了坏死、有丝分裂、分化程度、手术切缘以及是否存在神经纤维瘤成分。
21例患者中,男性15例,女性6例,年龄9 - 86岁。7例与神经纤维瘤病相关,14例为孤立性MPNST。合并神经纤维瘤病的患者比孤立性MPNST患者年轻(诊断时平均年龄32岁 vs 60.2岁)。前一组患者的肿瘤倾向于位于中央。在两名复发肿瘤患者中发现了间变改变,这两名患者均死于该疾病。21例病例中,光学显微镜下分别在两个肿瘤中观察到上皮样分化和骨样化生。患有冯雷克林霍增氏病且肿瘤巨大、切除不完全的患者预后最差。在5例随访超过两年的神经纤维瘤病患者中,仅1例存活且未发现复发。其余4例患者在3 - 9个月内死亡。然而,14例孤立性MPNST患者中,仅2例死于该疾病,其中10例存活超过两年。
我们的研究表明,患有冯雷克林霍增氏病、肿瘤巨大且位于中央无法切除、伴有弥漫性间变改变的患者复发频繁且预后不良。建议采用手术切缘阴性的积极手术方法。当出现疼痛、快速生长的肿瘤时,应高度怀疑恶性转化。