Nijssen P C, Deprez R H, Tijssen C C, Hagemeijer A, Arnoldus E P, Teepen J L, Holl R, Niermeyer M F
Department of Neurology, St Elisabeth Hospital, Tilburg, The Netherlands.
J Neurol Neurosurg Psychiatry. 1994 Oct;57(10):1245-8. doi: 10.1136/jnnp.57.10.1245.
A family with anaplastic ependymomas, histologically verified in three cases and neuroradiologically suggested in a fourth, is presented. Two healthy brothers both had two affected sons. All four male patients were younger than 5 years at the time of diagnosis. Two boys died before the age of 3 years. Genotype analysis (with polymorphic DNA markers for chromosome 22 and interphase cytogenetic analysis) of one of the tumours showed a subpopulation of tumour cells with monosomy of (part of) chromosome 22. Non-neoplastic cells of this patient showed a normal karyotype. These findings give further evidence for the role of a tumour suppressor gene on chromosome 22 in the pathogenesis of familial ependymal tumours.
本文报告了一个患有间变性室管膜瘤的家族,其中3例经组织学证实,第4例经神经放射学提示。两个健康的兄弟都有两个患病的儿子。所有4名男性患者在诊断时均未满5岁。其中一名肿瘤患者的基因型分析(使用22号染色体的多态性DNA标记和间期细胞遗传学分析)显示,肿瘤细胞亚群存在(部分)22号染色体单体性。该患者的非肿瘤细胞显示核型正常。这些发现进一步证明了22号染色体上的肿瘤抑制基因在家族性室管膜瘤发病机制中的作用。