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双等位基因MSH2突变引起的家族性T细胞非霍奇金淋巴瘤。

Familial T-cell non-Hodgkin lymphoma caused by biallelic MSH2 mutations.

作者信息

Scott Richard H, Homfray Tessa, Huxter Nicola L, Mitton Sally G, Nash Ruth, Potter Mike N, Lancaster Donna, Rahman Nazneen

机构信息

Section of Cancer Genetics, Institute of Cancer Research, 15 Cotswold Road, Sutton, Surrey, UK, SM2 5NG.

出版信息

J Med Genet. 2007 Jul;44(7):e83. doi: 10.1136/jmg.2007.048942.

Abstract

Familial non-Hodgkin lymphoma (NHL) is rare and in most cases, no underlying cause is identifiable. We report homozygous truncating mutations in the mismatch repair gene MSH2 (226C-->T; Q76X) in three siblings who each developed T-cell NHL in early childhood. All three children had hyperpigmented and hypopigmented skin lesions. Constitutional biallelic MSH2 mutations have previously been reported in five individuals, all of whom developed malignancy in childhood. Familial lymphoma has not been reported in this context or in association with biallelic mutations in the other mismatch repair genes MLH1, MSH6 or PMS2. In addition, hypopigmented skin lesions have not previously been reported in biallelic MSH2 carriers. Our findings therefore expand the spectrum of phenotypes associated with biallelic MSH2 mutations and identify a new cause of familial lymphoma. Moreover, the diagnosis has important management implications as it allows the avoidance of chemotherapeutic agents likely to be ineffective and mutagenic in the proband, and the provision of cascade genetic testing and tumour screening for relatives.

摘要

家族性非霍奇金淋巴瘤(NHL)较为罕见,在大多数情况下,无法确定潜在病因。我们报告了三个同胞中错配修复基因MSH2(226C→T;Q76X)的纯合截断突变,这三个同胞均在幼儿期患T细胞NHL。所有三个孩子都有色素沉着过度和色素沉着不足的皮肤病变。此前已报道五例个体存在双等位基因MSH2突变,他们均在儿童期发生恶性肿瘤。在这种情况下,尚未有家族性淋巴瘤的报道,也未发现与其他错配修复基因MLH1、MSH6或PMS2的双等位基因突变有关。此外,此前尚未报道双等位基因MSH2携带者出现色素沉着不足的皮肤病变。因此,我们的研究结果扩展了与双等位基因MSH2突变相关的表型谱,并确定了家族性淋巴瘤的一个新病因。此外,该诊断具有重要的管理意义,因为它可以避免使用可能对先证者无效且具有致突变性的化疗药物,并为亲属提供级联基因检测和肿瘤筛查。

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