Genomics Medicine Unit, Navarrabiomed, Pamplona, Spain.
Department of Pathology, Belfast Health and Social Care Trust, Belfast, UK.
Genes Chromosomes Cancer. 2021 Mar;60(3):217-233. doi: 10.1002/gcc.22913. Epub 2020 Nov 19.
Gynecologic sarcomas are uncommon neoplasms, the majority occurring in the uterus. Due to the diverse nature of these, the description of "new" morphological types and the rarity of some of them, pathological diagnosis and treatment is often challenging. Finding genetic alterations specific to, and frequently occurring, in a certain type can aid in the diagnosis. DICER1 is a highly conserved ribonuclease crucial in the biogenesis of microRNAs and mutations in DICER1 (either somatic or germline) have been detected in a wide range of sarcomas including genitourinary embryonal rhabdomyosarcomas (ERMS) and adenosarcomas. Importantly, DICER1-associated sarcomas share morphological features irrespective of the site of origin such that the pathologist can strongly suspect a DICER1 association. A review of the literature shows that almost all gynecologic ERMS reported (outside of the vagina) harbor DICER1 alterations, while approximately 20% of adenosarcomas also do so. These two tumor types exhibit significant morphological overlap and DICER1 tumor testing may be helpful in distinguishing between them, because a negative result makes ERMS unlikely. Given that germline pathogenic DICER1 variants are frequent in uterine (corpus and cervix) ERMS and pathogenic germline variants in this gene cause a hereditary cancer predisposition syndrome (DICER1 syndrome), patients diagnosed with these neoplasms should be referred to medical genetic services. Cooperation between pathologists and geneticists is crucial and will help in improving the diagnosis and management of these uncommon sarcomas.
妇科肉瘤是罕见的肿瘤,大多数发生在子宫。由于这些肿瘤的多样性,以及某些肿瘤的形态学类型的描述和罕见性,病理诊断和治疗常常具有挑战性。发现特定于某种类型且经常发生的遗传改变可以辅助诊断。DICER1 是一种高度保守的核糖核酸酶,对 microRNAs 的生物发生至关重要,在包括泌尿生殖系胚胎横纹肌肉瘤(ERMS)和腺肉瘤在内的广泛肉瘤中已经检测到 DICER1 突变(体细胞或种系)。重要的是,与 DICER1 相关的肉瘤无论起源部位如何,其形态特征都相似,因此病理学家可以强烈怀疑与 DICER1 相关。对文献的回顾表明,几乎所有报道的妇科 ERMS(阴道外)都存在 DICER1 改变,而大约 20%的腺肉瘤也是如此。这两种肿瘤类型具有显著的形态学重叠,DICER1 肿瘤检测可能有助于区分它们,因为阴性结果使 ERMS 不太可能。鉴于种系致病性 DICER1 变体在子宫(体部和颈部)ERMS 中频繁发生,并且该基因中的致病性种系变体导致遗传性癌症易感性综合征(DICER1 综合征),因此应将诊断出这些肿瘤的患者转介至医学遗传服务。病理学家和遗传学家之间的合作至关重要,将有助于改善这些罕见肉瘤的诊断和管理。