Department of Oncology, Sidney Kimmel Comprehensive Cancer Center, School of Medicine, Johns Hopkins University, 1650 Orleans St, Baltimore, MD, 21231, USA.
Johns Hopkins All Children's Hospital, 600 5th St. South, St. Petersburg, FL, 33701, USA.
Acta Neuropathol Commun. 2022 Feb 24;10(1):25. doi: 10.1186/s40478-022-01326-7.
Medulloblastoma (MB) is the most common malignant brain tumor in children. There remains an unmet need for diagnostics to sensitively detect the disease, particularly recurrences. Cerebrospinal fluid (CSF) provides a window into the central nervous system, and liquid biopsy of CSF could provide a relatively non-invasive means for disease diagnosis. There has yet to be an integrated analysis of the transcriptomic, metabolomic, and lipidomic changes occurring in the CSF of children with MB. CSF samples from patients with (n = 40) or without (n = 11; no cancer) MB were subjected to RNA-sequencing and high-resolution mass spectrometry to identify RNA, metabolite, and lipid profiles. Differentially expressed transcripts, metabolites, and lipids were identified and their biological significance assessed by pathway analysis. The DIABLO multivariate analysis package (R package mixOmics) was used to integrate the molecular changes characterizing the CSF of MB patients. Differentially expressed transcripts, metabolites, and lipids in CSF were discriminatory for the presence of MB but not the exact molecular subtype. One hundred and ten genes and ten circular RNAs were differentially expressed in MB CSF compared with normal, representing TGF-β signaling, TNF-α signaling via NF-kB, and adipogenesis pathways. Tricarboxylic acid cycle and other metabolites (malate, fumarate, succinate, α-ketoglutarate, hydroxypyruvate, N-acetyl-aspartate) and total triacylglycerols were significantly upregulated in MB CSF compared with normal CSF. Although separating MBs into subgroups using transcriptomic, metabolomic, and lipid signatures in CSF was challenging, we were able to identify a group of omics signatures that could separate cancer from normal CSF. Metabolic and lipidomic profiles both contained indicators of tumor hypoxia. Our approach provides several candidate signatures that deserve further validation, including the novel circular RNA circ_463, and insights into the impact of MB on the CSF microenvironment.
髓母细胞瘤(MB)是儿童中最常见的恶性脑肿瘤。目前仍需要开发诊断方法来灵敏地检测这种疾病,尤其是复发。脑脊液(CSF)为中枢神经系统提供了一个窗口,而 CSF 的液体活检可能为疾病诊断提供一种相对非侵入性的手段。目前尚未对 MB 患儿 CSF 中发生的转录组、代谢组和脂质组变化进行综合分析。对来自患有 MB 患者(n = 40)或无 MB 患者(n = 11;无癌症)的 CSF 样本进行了 RNA-seq 和高分辨率质谱分析,以鉴定 RNA、代谢物和脂质谱。通过通路分析鉴定差异表达的转录物、代谢物和脂质,并评估其生物学意义。使用 DIABLO 多变量分析包(R 包 mixOmics)整合了表征 MB 患者 CSF 的分子变化。CSF 中的差异表达转录物、代谢物和脂质可区分 MB 的存在,但不能区分确切的分子亚型。与正常 CSF 相比,MB CSF 中 110 个基因和 10 个环状 RNA 表达差异,代表 TGF-β 信号、TNF-α 通过 NF-kB 的信号和脂肪生成途径。三羧酸循环和其他代谢物(苹果酸、延胡索酸、琥珀酸、α-酮戊二酸、羟丙酮酸、N-乙酰天冬氨酸)和总三酰甘油在 MB CSF 中明显上调,而在正常 CSF 中则明显下调。尽管使用 CSF 中的转录组、代谢组和脂质组学特征将 MB 分组为亚组具有挑战性,但我们能够鉴定出一组可以将癌症与正常 CSF 区分开的组学特征。代谢组学和脂质组学图谱均包含肿瘤缺氧的指标。我们的方法提供了一些值得进一步验证的候选特征,包括新型环状 RNA circ_463,以及深入了解 MB 对 CSF 微环境的影响。