Departments of Laboratory Medicine and Pathology.
Radiology.
Am J Surg Pathol. 2021 Aug 1;45(8):1082-1090. doi: 10.1097/PAS.0000000000001690.
Posterior fossa (PF) diffuse gliomas in pediatric patients frequently harbor the H3 K27M mutation. Among adults, PF diffuse gliomas are rare, with limited data regarding molecular features and clinical outcomes. We identified 28 adult PF diffuse glioma patients (17 males; median: 50 y, range: 19 to 78 y), with surgery performed at our institution (13 brainstem; 15 cerebellum). Histologic subtypes included anaplastic astrocytoma (n=21), glioblastoma (n=6), and diffuse astrocytoma (n=1). Immunohistochemistry was performed for H3 K27M (n=26), IDH1-R132H (n=28), and ATRX (n=28). A 150-gene neuro-oncology-targeted next-generation sequencing panel was attempted in 24/28, with sufficient informative material in 15 (51.7%). Tumors comprised 4 distinct groups: driver mutations in H3F3A (brainstem=4; cerebellum=2), IDH1 (brainstem=4; cerebellum=4), TERT promotor mutation (brainstem=0; cerebellum=3), and none of these (n=5), with the latter harboring mutations of TP53, PDGFRA, ATRX, NF1, and RB1. All TERT promoter-mutant cases were IDH-wild-type and arose within the cerebellum. To date, 20 patients have died of disease, with a median survival of 16.3 months, 1-year survival of 67.5%. Median survival within the subgroups included: H3F3A=16.4 months, IDH mutant=113.4 months, and TERT promoter mutant=12.9 months. These findings suggest that PF diffuse gliomas affecting adults show molecular heterogeneity, which may be associated with patient outcomes and possible response to therapy, and supports the utility of molecular testing in these tumors.
儿童患者的后颅窝(PF)弥漫性神经胶质瘤常携带 H3 K27M 突变。在成年人中,PF 弥漫性神经胶质瘤较为罕见,关于其分子特征和临床结局的数据有限。我们鉴定了 28 名成年 PF 弥漫性神经胶质瘤患者(男性 17 名;中位年龄 50 岁,范围 19 至 78 岁),这些患者均在我院接受了手术(脑干 13 例,小脑 15 例)。组织学亚型包括间变性星形细胞瘤(n=21)、胶质母细胞瘤(n=6)和弥漫性星形细胞瘤(n=1)。免疫组化检测 H3 K27M(n=26)、IDH1-R132H(n=28)和 ATRX(n=28)。我们尝试对 28 例中的 24 例进行了 150 个神经肿瘤靶向的下一代测序面板检测,其中 15 例(51.7%)有足够的信息材料。肿瘤分为 4 个不同的组:H3F3A 中的驱动突变(脑干=4;小脑=2)、IDH1(脑干=4;小脑=4)、TERT 启动子突变(脑干=0;小脑=3)和无这些突变(n=5),后者存在 TP53、PDGFRA、ATRX、NF1 和 RB1 的突变。所有 TERT 启动子突变病例均为 IDH 野生型,且发生于小脑内。迄今为止,20 名患者死于疾病,中位生存期为 16.3 个月,1 年生存率为 67.5%。各亚组的中位生存期包括:H3F3A=16.4 个月,IDH 突变型=113.4 个月,TERT 启动子突变型=12.9 个月。这些发现表明,影响成年人的 PF 弥漫性神经胶质瘤显示出分子异质性,这可能与患者的结局和可能的治疗反应相关,并支持对这些肿瘤进行分子检测。