Yuqi Miao, Di Wu, Yu Li, Yangmingyue Ji, Yanmei Sang
{"title":"Radiation therapy for childhood-onset craniopharyngioma: systematic review and meta-analysis.","authors":"Yuqi Miao, Di Wu, Yu Li, Yangmingyue Ji, Yanmei Sang","doi":"10.1007/s11060-024-04914-w","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>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.</p><p><strong>Methods: </strong>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.</p><p><strong>Results: </strong>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.</p><p><strong>Conclusion: </strong>Radiotherapy is a suitable adjuvant or alternative treatment method for childhood CP patients. However, patients inevitably face significant long-term treatment-related complications.</p>","PeriodicalId":16425,"journal":{"name":"Journal of Neuro-Oncology","volume":" ","pages":"89-98"},"PeriodicalIF":3.2000,"publicationDate":"2025-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Neuro-Oncology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s11060-024-04914-w","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/1/3 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0
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.
背景:颅咽管瘤(CP)是一种起源于鞍区Rathke袋残余的良性肿瘤,约占所有颅咽管瘤病例的30%。放射疗法用于治疗CP患者已有几十年的历史;然而,目前仍缺乏对儿童CP患者接受外放射治疗的长期肿瘤控制结果的系统评价。方法:我们对多个数据库进行了全面的检索,以研究接受外部放疗的儿童期发病CP(COCP)患者的肿瘤进展率。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。meta回归用于探讨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)随访时间为2至14.9年。合并总体肿瘤进展率为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)。meta回归显示以下潜在危险因素均未显著影响复发率的异质性:放射方式(光子vs质子),中位(平均)随访时间,或未行手术切除的患者比例。生长激素缺乏症(GHD)、甲状腺激素缺乏症(THD)、促肾上腺皮质激素缺乏症(ACTHD)、促性腺激素释放激素缺乏症(GnRHD)和尿囊症(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)。血管病变的总发病率为0.06 (95% CI 0.04-0.09),光子和质子治疗的发病率相似。结论:放射治疗是儿童CP患者的一种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。
期刊介绍:
The Journal of Neuro-Oncology is a multi-disciplinary journal encompassing basic, applied, and clinical investigations in all research areas as they relate to cancer and the central nervous system. It provides a single forum for communication among neurologists, neurosurgeons, radiotherapists, medical oncologists, neuropathologists, neurodiagnosticians, and laboratory-based oncologists conducting relevant research. The Journal of Neuro-Oncology does not seek to isolate the field, but rather to focus the efforts of many disciplines in one publication through a format which pulls together these diverse interests. More than any other field of oncology, cancer of the central nervous system requires multi-disciplinary approaches. To alleviate having to scan dozens of journals of cell biology, pathology, laboratory and clinical endeavours, JNO is a periodical in which current, high-quality, relevant research in all aspects of neuro-oncology may be found.