Primary Gamma Knife Radiosurgery for pineal region tumors: A systematic review and pooled analysis of available literature with histological stratification

IF 8.3 1区 医学 Q1 ENDOCRINOLOGY & METABOLISM Journal of Pineal Research Pub Date : 2023-09-13 DOI:10.1111/jpi.12910
Filippo Gagliardi, Pierfrancesco De Domenico, Enrico Garbin, Silvia Snider, Pietro Mortini
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Abstract

Pineal region tumors (PTs) represent extremely rare pathologies, characterized by highly heterogeneous histological patterns. Most of the available evidence for Gamma Knife radiosurgical (GKSR) treatment of PTs arises from multimodal regimens, including GKSR as an adjuvant modality or as a salvage treatment at recurrence. We aimed to gather existing evidence on the topic and analyze single-patient-level data to address the efficacy and safety of primary GKSR. This is a systematic review of the literature (PubMed, Embase, Cochrane, Science Direct) and pooled analysis of single-patient-level data. A total of 1054 original works were retrieved. After excluding duplicates and irrelevant works, we included 13 papers (n = 64 patients). An additional 12 patients were included from the authors' original series. A total of 76 patients reached the final analysis; 56.5% (n = 43) received a histological diagnosis. Confirmed lesions included pineocytoma WHO grade I (60.5%), pineocytoma WHO grade II (14%), pineoblastoma WHO IV (7%), pineal tumor with intermediate differentiation WHO II/III (4.7%), papillary tumor of pineal region WHO II/III (4.7%), germ cell tumor (2.3%), neurocytoma WHO I (2.3%), astrocytoma WHO II (2.3%) and WHO III (2.3%). Presumptive diagnoses were achieved in the remaining 43.5% (n = 33) of cases and comprised of pineocytoma (9%), germ cell tumor (6%), low-grade glioma (6%), high-grade glioma (3%), meningioma (3%) and undefined in 73%. The mean age at the time of GKSR was 38.7 years and the mean lesional volume was 4.2 ± 4 cc. All patients received GKSR with a mean marginal dose of 14.7 ± 2.1 Gy (50% isodose). At a median 36-month follow-up, local control was achieved in 80.3% of cases. Thirteen patients showed progression after a median time of 14 months. Overall mortality was 13.2%. The median OS was not reached for all included lesions, except high-grade gliomas (8mo). The 3-year OS was 100% for LGG and pineal tumors with intermediate differentiation, 91% for low-grade pineal lesions, 66% for high-grade pineal lesions, 60% for germ cell tumors (GCTs), 50% for HGG, and 82% for undetermined tumors. The 3-year progression-free survival (PFS) was 100% for LGG and pineal intermediate tumors, 86% for low-grade pineal, 66% for high-grade pineal, 33.3% for GCTs, and 0% for HGG. Median PFS was 5 months for HGG and 34 months for GCTs. The radionecrosis rate was 6%, and cystic degeneration was observed in 2%. Ataxia as a presenting symptom strongly predicted mortality (odds ratio [OR] 104, p = .02), while GCTs and HGG histology well predicted PD (OR: 13, p = .04). These results support the efficacy and safety of primary GKSR treatment of PTs. Further studies are needed to validate these results, which highlight the importance of the initial presumptive diagnosis for choosing the best therapeutic strategy.

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松果体区肿瘤的原发性伽玛刀放射治疗:一项系统综述和组织学分层的文献汇总分析
松果体区域肿瘤(PT)代表了极其罕见的病理,其特征是高度异质的组织学模式。伽玛刀放射外科(GKSR)治疗PT的大多数可用证据来自多模式方案,包括GKSR作为辅助模式或复发时的挽救治疗。我们旨在收集有关该主题的现有证据,并分析单个患者水平的数据,以解决原发性GKSR的疗效和安全性问题。这是对文献(PubMed、Embase、Cochrane、Science Direct)和单患者水平数据的汇总分析的系统综述。共检索到1054件原创作品。在排除重复和无关作品后,我们纳入了13篇论文(n = 64名患者)。另外12名患者来自作者的原始系列。共有76名患者达到最终分析;56.5%(n = 43)接受组织学诊断。已确诊的病变包括世界卫生组织I级(60.5%)、世界卫生组织II级(14%)、世界卫生组织IV级(7%)、中分化松果体瘤世界卫生组织II/III(4.7%)、松果体区乳头状瘤世界卫生组织II/III(4.7%)、生殖细胞瘤(2.3%)、世界卫生组织I级(2.3%)神经细胞瘤、星形细胞瘤世界卫生组织II(2.3%)和世界卫生组织III(2.3%)。其余43.5%(n = 33)例,包括松果细胞瘤(9%)、生殖细胞瘤(6%)、低级别神经胶质瘤(6%。GKSR时的平均年龄为38.7岁,平均病变体积为4.2 ± 4毫升。所有患者均接受GKSR,平均边际剂量为14.7 ± 2.1 Gy(50%等剂量)。在中位36个月的随访中,80.3%的病例实现了局部控制。13名患者在中位时间14个月后出现进展。总死亡率为13.2%。除高级别胶质瘤(8mo)外,所有纳入的病变均未达到中位OS。LGG和中等分化的松果体肿瘤的3年OS为100%,低级别松果体病变为91%,高级别松果体损伤为66%,生殖细胞肿瘤(GCTs)为60%,HGG为50%,未确定肿瘤为82%。LGG和松果体中间肿瘤的3年无进展生存率(PFS)为100%,低度松果体为86%,高度松果体为66%,GCT为33.3%,HGG为0%。HGG的中位PFS为5个月,GCT为34个月。放射性坏死率为6%,囊性变性率为2%。共济失调作为一种表现症状有力地预测了死亡率(比值比[OR]104,p = .02),而GCT和HGG组织学很好地预测了PD(OR:13,p = .04)。这些结果支持GKSR治疗PT的有效性和安全性。需要进一步的研究来验证这些结果,这些结果强调了初步推定诊断对选择最佳治疗策略的重要性。
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来源期刊
Journal of Pineal Research
Journal of Pineal Research 医学-内分泌学与代谢
CiteScore
17.70
自引率
4.90%
发文量
66
审稿时长
1 months
期刊介绍: The Journal of Pineal Research welcomes original scientific research on the pineal gland and melatonin in vertebrates, as well as the biological functions of melatonin in non-vertebrates, plants, and microorganisms. Criteria for publication include scientific importance, novelty, timeliness, and clarity of presentation. The journal considers experimental data that challenge current thinking and welcomes case reports contributing to understanding the pineal gland and melatonin research. Its aim is to serve researchers in all disciplines related to the pineal gland and melatonin.
期刊最新文献
Cover image RETRACTION: Effects of Melatonin on Fatty Liver Disease: The Role of NR4A1/DNA-Pkcs/P53 Pathway, Mitochondrial Fission, and Mitophagy From Seasonality to Species Conservation: Chronobiological Research on European Hamsters in Strasbourg, France Melatonin Inhibits ET-1 Production to Break Crosstalk Between Prostate Cancer and Bone Cells: Implication for Osteoblastic Bone Metastasis Treatment Issue Information
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