Rubin B P, Chen C J, Morgan T W, Xiao S, Grier H E, Kozakewich H P, Perez-Atayde A R, Fletcher J A
Department of Pathology, Brigham and Women's Hospital, Boston, Massachusetts 02115, USA.
Am J Pathol. 1998 Nov;153(5):1451-8. doi: 10.1016/S0002-9440(10)65732-X.
Morphological, cytogenetic, and biological evidence supports a relationship between congenital (infantile) fibrosarcoma (CFS) and congenital mesoblastic nephroma (CMN). These tumors have a very similar histological appearance, and they are both associated with polysomies for chromosomes 8, 11, 17, and 20. Recently, CFS was shown to contain a novel t(12; 15)(p13;q25) translocation resulting in ETV6-NTRK3 gene fusion. The aims of this study were to determine whether congenital mesoblastic nephroma contains the t(12;15)(p13;q25) translocation and ETV6-NTRK3 gene fusion and whether ETV6-NTRK3 fusions, in CMN and CFS, antedate acquisition of nonrandom chromosome polysomies. To address these aims, we evaluated 1) ETV6-NTRK3 fusion transcripts by reverse transcriptase polymerase chain reaction and sequence analysis, 2) genomic ETV6-region chromosomal rearrangement by fluorescence in situ hybridization, and 3) chromosomal polysomies by karyotyping and fluorescence in situ hybridization. We report ETV6-NTRK3 fusion transcripts and/or ETV6-region rearrangement in five of six CMNs and in five of five CFSs. The ETV6-NTRK3 fusion transcripts and/or ETV-region chromosome rearrangements were demonstrated in two CMNs and one CFS that lacked chromosome polysomies. These findings demonstrate that t(12;15) translocation, and the associated ETV6-NTRK3 fusion, can antedate acquisition of chromosome polysomies in CMN and CFS. CMN and CFS are pathogenetically related, and it is likely that they represent a single neoplastic entity, arising in either renal or soft tissue locations.
形态学、细胞遗传学和生物学证据支持先天性(婴儿型)纤维肉瘤(CFS)与先天性中胚层肾瘤(CMN)之间存在关联。这些肿瘤具有非常相似的组织学外观,并且都与8号、11号、17号和20号染色体的多体性相关。最近研究表明,CFS含有一种新的t(12; 15)(p13;q25)易位,导致ETV6-NTRK3基因融合。本研究的目的是确定先天性中胚层肾瘤是否含有t(12;15)(p13;q25)易位和ETV6-NTRK3基因融合,以及在CMN和CFS中,ETV6-NTRK3融合是否早于非随机染色体多体性的出现。为了实现这些目标,我们进行了以下评估:1)通过逆转录聚合酶链反应和序列分析检测ETV6-NTRK3融合转录本;2)通过荧光原位杂交检测基因组ETV6区域的染色体重排;3)通过核型分析和荧光原位杂交检测染色体多体性。我们报告在6例CMN中的5例以及5例CFS中的5例中发现了ETV6-NTRK3融合转录本和/或ETV6区域重排。在2例CMN和1例CFS中,尽管缺乏染色体多体性,但仍检测到了ETV6-NTRK3融合转录本和/或ETV区域染色体重排。这些发现表明,t(12;15)易位以及相关的ETV6-NTRK3融合可能早于CMN和CFS中染色体多体性的出现。CMN和CFS在发病机制上相关,它们可能代表了一种单一的肿瘤实体,起源于肾脏或软组织部位。