Department of Pathology, Indiana University, Indianapolis, IN, USA.
Solid Tumor Molecular Pathology Laboratory, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Bologna, Italy.
Histopathology. 2024 Jul;85(1):75-80. doi: 10.1111/his.15175. Epub 2024 Mar 26.
Testicular Leydig cell tumours (LCTs) are the most common type of sex cord-stromal tumour in men, representing 1%-3% of all testicular neoplasms. Among testicular sex cord-stromal tumours, CTNNB1 mutations and nuclear expression of β-catenin have been typically associated with Sertoli cell tumour. Recent genomic analyses have shown that CTNNB1 variants are also identified in a subset of LCTs; however, the frequency and clinicopathologic associations of β-catenin alterations remain incompletely understood in this tumour type.
In this study, we evaluated 32 LCTs (five malignant/metastasizing, 27 nonmetastasizing) using β-catenin immunohistochemistry and DNA sequencing.
Immunohistochemistry revealed focal or multifocal nuclear β-catenin expression in 47% of the tumours. Diffuse nuclear β-catenin expression (in >50% of the tumour cells) was not detected in any of the cases analysed herein. Comparison of β-catenin-positive and β-catenin-negative cases did not show significant differences in the frequency of adverse histopathologic findings or malignant clinical behaviour. DNA sequencing performed de novo on a subset of seven cases revealed the presence of exon 3 CTNNB1 variants in four of them (4/7, 57%), with variant allele frequencies (VAF) ranging from 7 to 33%. Two additional β-catenin-positive cases that had been sequenced as part of a previous study harboured exon 3 CTNNB1 variants at VAF of 28% and 7%, respectively.
These results demonstrate that β-catenin alterations are relatively common in LCT, most likely occurring as subclonal events that are not enriched in cases with aggressive features. Further studies are needed to clarify the oncogenic role of β-catenin in this tumour type.
睾丸间质细胞瘤(LCT)是男性中最常见的性索-间质肿瘤类型,占所有睾丸肿瘤的 1%-3%。在睾丸性索-间质肿瘤中,CTNNB1 突变和β-连环蛋白的核表达通常与支持细胞瘤相关。最近的基因组分析表明,CTNNB1 变体也存在于部分 LCT 中;然而,在这种肿瘤类型中,β-连环蛋白改变的频率和临床病理关联仍不完全清楚。
本研究使用β-连环蛋白免疫组化和 DNA 测序评估了 32 例 LCT(5 例恶性/转移,27 例非转移)。
免疫组化显示 47%的肿瘤存在局灶或多灶性核β-连环蛋白表达。在本研究分析的任何病例中均未检测到弥漫性核β-连环蛋白表达(>50%的肿瘤细胞)。β-连环蛋白阳性和β-连环蛋白阴性病例之间的比较显示,不良组织病理学发现或恶性临床行为的频率无显著差异。对 7 例病例中的一部分进行的从头 DNA 测序显示,其中 4 例(4/7,57%)存在 CTNNB1 外显子 3 变体,变异等位基因频率(VAF)范围为 7%至 33%。另外 2 例β-连环蛋白阳性病例,在之前的研究中测序时,分别携带 CTNNB1 外显子 3 变体,VAF 分别为 28%和 7%。
这些结果表明,β-连环蛋白改变在 LCT 中较为常见,很可能是亚克隆事件,在具有侵袭性特征的病例中并不富集。需要进一步研究来阐明β-连环蛋白在这种肿瘤类型中的致癌作用。