Martin Paloma, Santón Almudena, García-Cosio Mónica, Bellas Carmen
Laboratory of Molecular Pathology, Department of Pathology, Hospital Universitario Puerta de Hierro, Madrid, Spain.
Mod Pathol. 2006 Jul;19(7):914-21. doi: 10.1038/modpathol.3800590. Epub 2006 Apr 7.
Monoclonal gammopathies are a group of disorders characterized by clonal proliferation and accumulation of immunoglobulin-producing plasma cells. Multiple myeloma and monoclonal gammopathy of undetermined significance are the most common monoclonal gammopathies; the two comprise a spectrum of disorders, ranging from a relatively benign disease, monoclonal gammopathy of undetermined significance, to a malignant disease, multiple myeloma. Aberrant promoter methylation represents a primary mechanism of gene silencing during tumorigenesis. DNA repair systems act to maintain genome integrity in the presence of replication errors, environmental insults, and the cumulative effects of aging. The methylation patterns of two genes implicated in DNA repair, O6 methylguanine DNA methyl-transferase (MGMT) and human mutL homologue1 (hMLH1) have been detected in various solid tumours. With the purpose of studying the gene silencing of MGMT and hMLH1 in plasma cell disorders, we investigated the methylation status and expression of both genes in: 29 cases of multiple myeloma; one case of plasma cell leukaemia; 13 cases of monoclonal gammopathy of undetermined significance; and two cases of polyclonal plasmacytosis, using methylation-specific polymerase-chain reaction and immunohistochemical techniques. Methylation frequencies for MGMT were 23% in multiple myeloma and 8% in monoclonal gammopathy of undetermined significance. It was 10% for hMLH1 in multiple myeloma. None of the patients diagnosed with monoclonal gammopathy of undetermined significance had hMLH1 hypermethylated. In addition, 50% of myeloma cases had a loss of hMLH1 expression, whereas silencing of MGMT was observed in 43% of myeloma and 36% of samples with monoclonal gammopathy of undetermined significance. This study indicates that repair pathway defects play a role in the pathogenesis and evolution of monoclonal gammopathies, and suggests that inactivation of hMLH1 could be implicated in multiple myeloma tumorigenesis.
单克隆丙种球蛋白病是一组以产生免疫球蛋白的浆细胞克隆性增殖和积聚为特征的疾病。多发性骨髓瘤和意义未明的单克隆丙种球蛋白病是最常见的单克隆丙种球蛋白病;这两种疾病构成了一系列病症,从相对良性的疾病——意义未明的单克隆丙种球蛋白病,到恶性疾病——多发性骨髓瘤。异常的启动子甲基化是肿瘤发生过程中基因沉默的主要机制。DNA修复系统在存在复制错误、环境损伤和衰老的累积效应时,起到维持基因组完整性的作用。在各种实体瘤中已检测到与DNA修复相关的两个基因,即O6甲基鸟嘌呤DNA甲基转移酶(MGMT)和人mutL同源物1(hMLH1)的甲基化模式。为了研究MGMT和hMLH1在浆细胞疾病中的基因沉默情况,我们采用甲基化特异性聚合酶链反应和免疫组化技术,调查了这两个基因在以下情况中的甲基化状态和表达:29例多发性骨髓瘤;1例浆细胞白血病;13例意义未明的单克隆丙种球蛋白病;以及2例多克隆浆细胞增多症。MGMT在多发性骨髓瘤中的甲基化频率为23%,在意义未明的单克隆丙种球蛋白病中为8%。hMLH1在多发性骨髓瘤中的甲基化频率为10%。诊断为意义未明的单克隆丙种球蛋白病的患者中,无一例hMLH1发生高甲基化。此外,50%的骨髓瘤病例存在hMLH1表达缺失,而在43%的骨髓瘤病例和36%的意义未明的单克隆丙种球蛋白病样本中观察到MGMT沉默。本研究表明,修复途径缺陷在单克隆丙种球蛋白病的发病机制和演变中起作用,并提示hMLH1失活可能与多发性骨髓瘤的肿瘤发生有关。