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使用下一代测序鉴定蕈样肉芽肿的致病变体。

Pathogenic variants of mycosis fungoides identified using next-generation sequencing.

作者信息

Shrestha Sunaina, Newsom Kimberly, Chaffin Joanna Melody, Seifert Robert P

机构信息

Department of Pathology, Immunology and Laboratory Medicine, University of Florida College of Medicine, Gainesville, FL, USA.

出版信息

J Hematop. 2024 Dec;17(4):209-214. doi: 10.1007/s12308-024-00607-5. Epub 2024 Sep 19.

Abstract

Mycosis fungoides (MF), the predominant form of cutaneous T-cell lymphoma (CTCL), poses diagnostic challenges due to its clinical and histological resemblance to benign skin disorders. Delayed diagnosis contributes to therapeutic delays, prompting exploration of advanced diagnostics tools. Next-generation sequencing (NGS) may enhance disease detection by identifying pathogenic variants common to CTCL but absent in benign inflammatory disorders. We aim to discuss novel and common pathogenic variants in CTCL to enhance the utility of NGS as a diagnostic adjunct. This pilot study employed (NGS) to identify pathogenic variants in 10 MF cases. Cases were selected based on PCR-confirmed T-cell receptor clonality, with adequate DNA for NGS. GatorSeq NGS Panel, Illumina NextSeq500, and QIAGEN Clinical Insight QCI software facilitated sequencing, analysis, and variant interpretation. NGS revealed eight novel mutations in genes including HLA-DRB1, AK2, ITPKB, HLA-B, TYRO3, and CHD2. Additionally, previously reported MF-associated mutations such as DNMT3A, STAT5B, and SOCS1 (mouse study only) were detected as well. Detected variants were involved in apoptotic, NF-kB, JAK-STAT, and TCR signaling pathways, providing insights into MF pathogenesis. Mutations in genes like APC, AK2, TYRO3, and ITPKB that regulate tumor proliferation and apoptosis were noted. MF cases were associated with HLA gene mutations. NGS may enhance MF diagnosis, as the detection of pathogenic variants, particularly those known to occur in MF, favors a neoplastic diagnosis over an inflammatory diagnosis. Continuing this work may lead to the discovery of therapeutic targets.

摘要

蕈样肉芽肿(MF)是皮肤T细胞淋巴瘤(CTCL)的主要形式,由于其临床和组织学表现与良性皮肤疾病相似,给诊断带来了挑战。诊断延迟会导致治疗延误,促使人们探索先进的诊断工具。下一代测序(NGS)可能通过识别CTCL中常见但在良性炎症性疾病中不存在的致病变异来提高疾病检测率。我们旨在讨论CTCL中的新型和常见致病变异,以提高NGS作为诊断辅助手段的效用。这项前瞻性研究采用NGS来鉴定10例MF病例中的致病变异。病例是根据PCR确认的T细胞受体克隆性选择的,有足够的DNA用于NGS。GatorSeq NGS Panel、Illumina NextSeq500和QIAGEN Clinical Insight QCI软件有助于测序、分析和变异解读。NGS在包括HLA-DRB1、AK2、ITPKB、HLA-B、TYRO3和CHD2等基因中发现了8个新突变。此外,还检测到了先前报道的与MF相关的突变,如DNMT3A、STAT5B和SOCS1(仅在小鼠研究中)。检测到的变异涉及凋亡、NF-κB、JAK-STAT和TCR信号通路,为MF发病机制提供了见解。注意到调节肿瘤增殖和凋亡的基因如APC、AK2、TYRO3和ITPKB中的突变。MF病例与HLA基因突变有关。NGS可能会提高MF的诊断率,因为致病变异的检测,特别是那些已知在MF中出现的变异,有利于肿瘤性诊断而非炎症性诊断。继续这项工作可能会导致治疗靶点的发现。

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