诊断沉默:非功能性甲状旁腺癌的分子景观。

IF 3.1 3区 医学 Q1 PATHOLOGY
Maaia Margo Jentus, Willem E Corver, Marieke Snel, Femke M van Haalen, Tom van Wezel, Dina Ruano, Ellen Kapiteijn, Stijn Crobach, Natasha M Appelman-Dijkstra, Abbey Schepers, Hans Morreau
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引用次数: 0

摘要

非功能性甲状旁腺癌(NFPC)是一种罕见的恶性肿瘤,自19世纪晚期甲状旁腺首次被描述以来,大约有50例报告。功能性甲状旁腺癌(PC)通常表现为严重甲状旁腺功能亢进症(HPT),可通过血清钙和甲状旁腺激素(PTH)水平监测,而NFPC缺乏生化标志物,仅通过影像学监测。在本研究中,我们报告了两例原发性NFPC的新病例,并提供了全面的文献回顾,包括免疫组织化学和分子检测的详细分析。我们还探讨了PC非功能状态的潜在机制。在一个病例中,通过免疫组化检测,肿瘤缺乏PTH的表达,但PTH mRNA的表达水平一直很低。排除了鉴别诊断,包括髓样和非髓样甲状腺癌、副神经节瘤和其他头颈部肿瘤。第二例显示斑片状残余甲状旁腺素免疫染色。两种肿瘤均表现出广泛的全染色体丢失,导致基因组接近单倍体,随后伴有(NFPC 2)或不伴有(NFPC 1)内重复/基因组加倍。两者的突变情况有所不同,均未显示CDC73突变或parafibromin表达缺失。在PTH免疫组化阴性的病例中,诊断原发性非功能性甲状旁腺癌尤其具有挑战性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Diagnosing the silent: the molecular landscape of non-functional parathyroid carcinoma.

Non-functional parathyroid carcinoma (NFPC) is an exceptionally rare malignancy, with approximately 50 cases reported since the parathyroid glands were first described in the late nineteenth century. Unlike functional parathyroid carcinomas (PC), which typically present with severe hyperparathyroidism (HPT) and are monitored via serum calcium and parathyroid hormone (PTH) levels, NFPC lacks biochemical markers and is monitored solely through imaging. In this study, we present two new cases of primary NFPC and provide a comprehensive review of the literature, including a detailed analysis of immunohistochemical and molecular testing. We also explore potential mechanisms underlying the non-functional state in PC. In one case, the tumor lacked PTH expression by immunohistochemistry, yet low levels of PTH mRNA expression were consistently detected. Differential diagnoses-including medullary and non-medullary thyroid carcinoma, paraganglioma, and other head and neck tumors-were excluded. The second case demonstrated patchy residual PTH immunostaining. Both tumors exhibited extensive whole chromosome losses resulting in a near-haploid genome, with (NFPC 2) or without (NFPC 1) subsequent endoreduplication/genome doubling. The mutational landscape differed between the two, with neither showing CDC73 mutations or loss of parafibromin expression. Diagnosing primary non-functional parathyroid carcinoma can be especially challenging in cases with negative PTH immunohistochemistry.

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来源期刊
Virchows Archiv
Virchows Archiv 医学-病理学
CiteScore
7.40
自引率
2.90%
发文量
204
审稿时长
4-8 weeks
期刊介绍: Manuscripts of original studies reinforcing the evidence base of modern diagnostic pathology, using immunocytochemical, molecular and ultrastructural techniques, will be welcomed. In addition, papers on critical evaluation of diagnostic criteria but also broadsheets and guidelines with a solid evidence base will be considered. Consideration will also be given to reports of work in other fields relevant to the understanding of human pathology as well as manuscripts on the application of new methods and techniques in pathology. Submission of purely experimental articles is discouraged but manuscripts on experimental work applicable to diagnostic pathology are welcomed. Biomarker studies are welcomed but need to abide by strict rules (e.g. REMARK) of adequate sample size and relevant marker choice. Single marker studies on limited patient series without validated application will as a rule not be considered. Case reports will only be considered when they provide substantial new information with an impact on understanding disease or diagnostic practice.
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