Tekkök Ismail H, Sav Aydin
Department of Neurosurgery, Mersin University School of Medicine, Zeytinlibahce Caddesi, 33079, Mersin, Turkey.
J Neurooncol. 2005 Jul;73(3):241-52. doi: 10.1007/s11060-004-5671-6.
This paper presents the case of an eight-year-old girl who presented with headache and vomiting and was found to harbor a right fronto-temporo-parietal, partially cystic and centrally solid tumor that measured 11 x 8 x 7 cm. This vascular tumor was gross totally removed. The initial histopathologic diagnosis was hemangiopericytoma and the patient received a total dose of 5330 cGy of external cranial radiation. Twelve months later, the patient presented with left lower quadrant pain and limping and the spinal MR scans showed metastases at T4-5, T7, T12-L1 and L3 levels. The voluminous lesion at T12-L1 was surgically removed. Histopathological examination of both specimens revealed that both tumors in fact were malignant rhabdoid tumor (MRT). The patient did not benefit from spinal surgery and died 4 months later. A review of the literature has shown that since Briner et al'. first report in 1985 [Pediatr Pathol 3: 117-118, 1985], 100 MRT cases have been published. More than two-thirds of reviewed cases presented with local recurrence or subarachnoid spread after a mean period of 6.9 months after diagnosis and died two months later. Infratentorial and pineal location and surgery limited to biopsy were poor prognostic indicators. Twenty-two cases remained alive at a mean period of 24.5 months. The longest survival with an intracranial MRT was 65 months. Of those remaining alive, 15 had no evidence of disease (NED). Our case is the first MRT case immunopositive for HMB-45 and has also shown that the MRT cells grow aggressive over time as demonstrated by a four-fold increase in MIB-1 labeling index.
本文介绍了一名8岁女孩的病例,该女孩出现头痛和呕吐症状,经检查发现患有一个位于右额颞顶叶的部分囊性、中央实性肿瘤,大小为11×8×7厘米。这个血管性肿瘤被完整切除。最初的组织病理学诊断为血管外皮细胞瘤,患者接受了总量为5330 cGy的颅外放射治疗。12个月后,患者出现左下腹疼痛和跛行,脊柱磁共振扫描显示在T4 - 5、T7、T12 - L1和L3水平有转移。T12 - L1处的巨大病变被手术切除。对两个标本的组织病理学检查显示,这两个肿瘤实际上都是恶性横纹肌样瘤(MRT)。患者未从脊柱手术中获益,4个月后死亡。文献回顾显示,自1985年Briner等人首次报告以来[《儿科病理学》3: 117 - 118, 1985],已发表了100例MRT病例。超过三分之二的回顾病例在诊断后平均6.9个月出现局部复发或蛛网膜下腔扩散,并在两个月后死亡。幕下和松果体部位以及仅限于活检的手术是不良预后指标。22例患者平均存活24.5个月。颅内MRT最长存活时间为65个月。在存活的患者中,15例无疾病证据(NED)。我们的病例是首例对HMB - 45免疫阳性的MRT病例,并且还表明,随着时间的推移,MRT细胞生长具有侵袭性,MIB - 1标记指数增加了四倍。