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2型神经纤维瘤病归因于临床正常母亲的生殖腺嵌合体,并鉴定出NF2基因的7种新突变。

Neurofibromatosis type 2 attributable to gonosomal mosaicism in a clinically normal mother, and identification of seven novel mutations in the NF2 gene.

作者信息

Sestini R, Vivarelli R, Balestri P, Ammannati F, Montali E, Papi L

机构信息

Department of Clinical Physiopathology, University of Florence, Italy.

出版信息

Hum Genet. 2000 Oct;107(4):366-71. doi: 10.1007/s004390000378.

Abstract

Neurofibromatosis type 2 (NF2) is an autosomal dominant cancer syndrome that predisposes to the development of bilateral vestibular schwannomas sometimes associated with schwannomas at other locations, meningiomas, ependymomas and juvenile posterior subcapsular lenticular opacities. This disease is caused by inactivating mutations in the NF2 tumour-suppressor gene, located in 22q12. Recently, somatic mosaicism has been demonstrated in some "de novo" NF2 patients. We here report the genetic study of 33 NF2 patients from 33 unrelated Italian families. Twelve mutations were characterised, including seven newly identified mutations and five recurrent ones. Furthermore, we describe one patient with an inactivating mutation that lies in exon 13 but that is present in only a portion of the lymphocytes and, more importantly, a clinically normal individual carrying a somatic/germinal mosaicism for a nonsense mutation in exon 10 of the NF2 gene. Our results confirm the relatively high percentage of mosaicism for mutations in the NF2 gene and establish the importance of evaluating genomic DNA from several tissues, in addition to lymphocytes, so as to identify mosaicism in "de novo" NF2 patients and their relatives. In addition, the demonstration of somatic and/or gonadal mosaicism is an important tool for accurate genetic counselling in families with sporadic cases of NF2.

摘要

2型神经纤维瘤病(NF2)是一种常染色体显性癌症综合征,易患双侧前庭神经鞘瘤,有时还伴有其他部位的神经鞘瘤、脑膜瘤、室管膜瘤和青少年后囊下晶状体混浊。这种疾病是由位于22q12的NF2肿瘤抑制基因的失活突变引起的。最近,在一些“新发”NF2患者中已证实存在体细胞镶嵌现象。我们在此报告对来自33个不相关意大利家庭的33例NF2患者的遗传学研究。鉴定出12种突变,包括7种新发现的突变和5种复发突变。此外,我们描述了一名患者,其失活突变位于外显子13,但仅存在于一部分淋巴细胞中,更重要的是,描述了一名临床正常个体,其携带NF2基因外显子10无义突变的体细胞/生殖细胞镶嵌现象。我们的结果证实了NF2基因突变镶嵌现象的相对高比例,并确立了除淋巴细胞外,评估多个组织的基因组DNA对于识别“新发”NF2患者及其亲属中镶嵌现象的重要性。此外,体细胞和/或性腺镶嵌现象的证实是对散发性NF2病例家庭进行准确遗传咨询的重要工具。

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