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儿童期颅咽管瘤的放射治疗:系统评价与荟萃分析

Radiation therapy for childhood-onset craniopharyngioma: systematic review and meta-analysis.

作者信息

Miao Yuqi, Wu Di, Li Yu, Ji Yangmingyue, Sang Yanmei

机构信息

Department of Endocrinology, Genetics and Metabolism, Beijing Children's Hospital, National Center for Children's Health, Capital Medical University, Beijing, 100045, China.

Beijing Key Laboratory for Genetics of Birth Defects, Beijing, 100045, China.

出版信息

J Neurooncol. 2025 Mar;172(1):89-98. doi: 10.1007/s11060-024-04914-w. Epub 2025 Jan 3.

Abstract

BACKGROUND

Craniopharyngioma (CP), a benign tumor originating from remnants of Rathke's pouch in the sellar region, accounts for approximately 30% of all cases of craniopharyngioma. Radiation therapy has been used to treat CP patients for decades; however, there is still a lack of systematic reviews on the long-term tumor control outcomes in pediatric CP patients treated with external radiation therapy.

METHODS

We conducted a comprehensive search of multiple databases for studies on the tumor progression rates of childhood-onset CP(COCP) patients who received external radiotherapy. We also recorded morbidities related to hypopituitarism and vasculopathy. A meta-analysis was performed to calculate the pooled incidence rates. Meta-regression was applied to explore potential sources of heterogeneity in the tumor progression rates.

RESULTS

A total of 22 studies were included after screening and eligibility assessment in accordance with PRISMA guidelines. The median (mean) follow-up period ranged from 2 to 14.9 years. The pooled overall tumor progression rate was 0.10 (95% CI 0.07-0.15). The recurrence rates were 0.14 (95% CI 0.09-0.19) for photon therapy and 0.04 (95% CI 0.01-0.07) for proton therapy. Meta-regression indicated that none of the following underlying risk factors significantly affected the heterogeneity of the recurrence rate: radiation modality (photon vs. proton), median (mean) follow-up duration, or the proportion of patients who did not undergo surgical resection. The pooled incidence of growth hormone deficiency (GHD), thyroid hormone deficiency (THD), adrenocorticotropic hormone deficiency (ACTHD), gonadotropin-releasing hormone deficiency (GnRHD), and diabetes insipidus (DI) were 0.81 (95% CI 0.70-0.90), 0.88 (95% CI 0.79-0.95), 0.69 (95% CI 0.52-0.85), 0.43 (95% CI 0.38-0.49), and 0.56 (95% CI 0.33-0.78), respectively. The pooled morbidity rate for vasculopathy was 0.06 (95% CI 0.04-0.09), with similar rates observed for both photon and proton therapy.

CONCLUSION

Radiotherapy is a suitable adjuvant or alternative treatment method for childhood CP patients. However, patients inevitably face significant long-term treatment-related complications.

摘要

背景

颅咽管瘤(CP)是一种起源于鞍区拉克氏囊残余组织的良性肿瘤,约占所有颅咽管瘤病例的30%。放射治疗已用于治疗CP患者数十年;然而,对于接受外照射治疗的小儿CP患者的长期肿瘤控制结果仍缺乏系统评价。

方法

我们对多个数据库进行了全面检索,以查找关于接受外照射放疗的儿童期发病CP(COCP)患者肿瘤进展率的研究。我们还记录了与垂体功能减退和血管病变相关的发病率。进行荟萃分析以计算合并发病率。应用荟萃回归来探索肿瘤进展率异质性的潜在来源。

结果

根据PRISMA指南进行筛选和资格评估后,共纳入22项研究。中位(平均)随访期为2至14.9年。合并的总体肿瘤进展率为0.10(95%CI 0.07 - 0.15)。光子治疗的复发率为0.14(95%CI 0.09 - 0.19),质子治疗的复发率为0.04(95%CI 0.01 - 0.07)。荟萃回归表明,以下潜在危险因素均未显著影响复发率的异质性:放疗方式(光子与质子)、中位(平均)随访持续时间或未接受手术切除的患者比例。生长激素缺乏(GHD)、甲状腺激素缺乏(THD)、促肾上腺皮质激素缺乏(ACTHD)、促性腺激素释放激素缺乏(GnRHD)和尿崩症(DI)的合并发病率分别为0.81(95%CI 0.70 - 0.90)、0.88(95%CI 0.79 - 0.95)、0.69(95%CI 0.52 - 0.85)、0.43(95%CI 0.38 - 0.49)和0.56(95%CI 0.33 - 0.78)。血管病变的合并发病率为0.06(95%CI 0.04 - 0.09),光子和质子治疗的发病率相似。

结论

放疗是小儿CP患者合适的辅助或替代治疗方法。然而,患者不可避免地面临显著的长期治疗相关并发症。

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