Department of Pathology, University Medical Center Groningen, Groningen, The Netherlands.
Department of Pathology, Shuang Ho Hospital, Taipei Medical University, Taipei, Taiwan.
Genes Chromosomes Cancer. 2019 Feb;58(2):100-110. doi: 10.1002/gcc.22681. Epub 2018 Oct 11.
Pediatric soft tissue tumors are relatively rare and show significant overlap in morphology and immunoprofile, often posing diagnostic and management challenges. Thus, their classification remains often subjective or lumped under "unclassified categories," as a number of lesions lack objective and reproducible criteria in diagnosis. Although in a subset of cases immunohistochemistry has been proved useful to identify a specific line of differentiation, most tumors lack a readily defined histogenesis, being characterized by a rather non-specific immunoprofile. Furthermore, tumors with an ambiguous diagnosis are difficult to grade and their risk of malignancy or clinical management remains uncertain. Advances in molecular genetics, including the more wide application of next generation sequencing in routine clinical practice, have improved diagnosis and refined classification based on objective molecular markers. Importantly, some soft tissue tumors in children are characterized by recurrent gene fusions involving either growth factors (eg, PDGFB) or protein kinases (eg, ALK, ROS, NTRK, BRAF), which have paved the way for new targeted treatments that block the respective upregulated downstream pathways. However, the majority of gene fusions or mutations detected in soft tissue tumors result in an abnormal function of transcription factors or chromatin remodeling. The present review focuses on the latest genetic discoveries in the spectrum of both benign and malignant pediatric soft tissue neoplasia. These genetic abnormalities promise to provide relevant insight for their proper classification, prognosis, and treatment. The entities discussed herein are grouped either based on their shared genetic mechanism or based on their presumed line of differentiation.
儿科软组织肿瘤相对少见,其形态学和免疫表型存在显著重叠,常导致诊断和管理方面的挑战。因此,它们的分类往往具有主观性,或者归入“未分类类别”,因为许多病变在诊断中缺乏客观和可重复的标准。尽管在一些病例中,免疫组织化学已被证明有助于确定特定的分化方向,但大多数肿瘤缺乏明确的组织发生,其免疫表型相当非特异性。此外,具有模糊诊断的肿瘤难以分级,其恶性肿瘤风险或临床管理仍然不确定。分子遗传学的进展,包括下一代测序在常规临床实践中的更广泛应用,提高了基于客观分子标志物的诊断和分类。重要的是,儿童中的一些软组织肿瘤具有涉及生长因子(例如 PDGFB)或蛋白激酶(例如 ALK、ROS、NTRK、BRAF)的复发性基因融合,为阻断各自上调的下游途径的新靶向治疗铺平了道路。然而,在软组织肿瘤中检测到的大多数基因融合或突变导致转录因子或染色质重塑的异常功能。本综述重点介绍了良性和恶性儿科软组织肿瘤谱中最新的遗传发现。这些遗传异常有望为其正确分类、预后和治疗提供相关见解。本文讨论的实体根据其共同的遗传机制或假定的分化方向进行分组。