Abstract
Introduction
Numerous studies have demonstrated Fractionated Stereotactic Radiotherapy's (FSRT) effectiveness in tumor control post-resection for craniopharyngiomas. Nevertheless, past literature has presented conflicting findings particularly regarding endocrine and visual function outcomes. This study aims to elucidate FSRT's efficacy and safety for this population.
Methods
Adhering to PRISMA, a systematic review and meta-analyses was conducted. Included studies had to report the effects of FSRT for treating craniopharyngiomas in a sample greater than four patients, addressing at least one of the outcomes of interest: improvement in visual acuity or field, new-onset hypopituitarism, effectiveness, and tumor progression. Relative risk with 95% confidence intervals were used to assess the outcomes.
Results
After retrieving a total of 1292 studies, 10 articles met the predefined criteria and thus were finally selected, amounting to a total of 256 patients. The improvement in visual acuity was estimated at 45% (95% CI: 6–83%), while the improvement in the visual field was 22% (95% CI: 0–51%). Regarding endocrine function, the new-onset hypopituitarism rate was found to be 5% (95% CI: 0–11%). Relative to FSRT effectiveness, the pooled estimate of the complete tumor response rate was 17% (95% CI: 4–30%), and the tumor progression rate was 7% (95% CI: 1–13%). Also, a 3-year progression-free survival rate of 98% (95% CI: 95–100%) was obtained.
Conclusion
Despite limitations and risks, FSRT shows promise as a viable therapeutic option for craniopharyngiomas, offering notable benefits for visual functions and tumor control. Further research is required to better understand the associated risks, benefits, and clinical utility.
Similar content being viewed by others
Data availability
No datasets were generated or analysed during the current study.
Abbreviations
- FSRT:
-
Fractionated Stereotactic Radiotherapy
- GTR:
-
Gross total resection
- NA:
-
Not Available
- PRISMA:
-
Preferred Reporting Items for Systematic Reviews and Meta-Analysis
- PFS:
-
Progression-Free Survival
- QOL:
-
Quality of life
- RT:
-
Radiation therapy
- ROBINS-I:
-
Risk Of Bias In Non-randomized Studies
- RR:
-
Relative Risk
References
Ostrom QT, Price M, Ryan K et al (2022) CBTRUS statistical report: Pediatric Brain Tumor Foundation childhood and adolescent primary brain and other central nervous system tumors diagnosed in the United States in 2014–2018. Neuro Oncol 24(Suppl 3):iii1–iii38. https://doi.org/10.1093/neuonc/noac161
Chou CL, Chen HH, Yang HC, et al. (2021) Effects of stereotactic radiosurgery versus conventional radiotherapy on body mass index in patients with craniopharyngioma. J Neurosurg Pediatr 28(1):43–49. Published 2021 May 14. https://doi.org/10.3171/2020.12.PEDS20165
Karavitaki N, Cudlip S, Adams CB, Wass JA (2006) Craniopharyngiomas. Endocr Rev 27(4):371–397. https://doi.org/10.1210/er.2006-0002
Ortiz Torres M, Shafiq I, Mesfin FB (2023) Craniopharyngioma. In: StatPearls. Treasure Island (FL): StatPearls Publishing
Müller HL, Merchant TE, Warmuth-Metz M, Martinez-Barbera JP, Puget S. (2019) Craniopharyngioma. Nat Rev Dis Primers 5(1):75. Published 2019 Nov 7. https://doi.org/10.1038/s41572-019-0125-9
Minniti G, Saran F, Traish D et al (2007) Fractionated stereotactic conformal radiotherapy following conservative surgery in the control of craniopharyngiomas. Radiother Oncol 82(1):90–95. https://doi.org/10.1016/j.radonc.2006.11.005
Selch MT, DeSalles AA, Wade M et al (2002) Initial clinical results of stereotactic radiotherapy for the treatment of craniopharyngiomas. Technol Cancer Res Treat 1(1):51–59. https://doi.org/10.1177/153303460200100107
Park HJ, Dho YS, Kim JH, Kim JW, Park CK, Kim YH (2020) Recurrence rate and prognostic factors for the adult craniopharyngiomas in long-term follow-up. World Neurosurg 133:e211–e217. https://doi.org/10.1016/j.wneu.2019.08.209
Astradsson A, MunckAfRosenschöld P, Feldt-Rasmussen U et al (2017) Visual outcome, endocrine function and tumor control after fractionated stereotactic radiation therapy of craniopharyngiomas in adults: findings in a prospective cohort. Acta Oncol 56(3):415–421. https://doi.org/10.1080/0284186X.2016.1270466
Roug S, Rasmussen AK, Juhler M et al (2010) Fractionated stereotactic radiotherapy in patients with acromegaly: an interim single-Centre audit. Eur J Endocrinol 162(4):685–694. https://doi.org/10.1530/EJE-09-1045
Nutting C, Brada M, Brazil L et al (1999) Radiotherapy in the treatment of benign meningioma of the skull base. J Neurosurg 90(5):823–827. https://doi.org/10.3171/jns.1999.90.5.0823
Guckenberger M, Baus WW, Blanck O, Combs SE, Debus J, Engenhart-Cabillic R, Gauer T, Grosu AL, Schmitt D, Tanadini-Lang S, Moustakis C (2020) Definition and quality requirements for stereotactic radiotherapy: consensus statement from the DEGRO/DGMP Working Group Stereotactic Radiotherapy and Radiosurgery Abstract Strahlentherapie und Onkologie 196(5):417–420. https://doi.org/10.1007/s00066-020-01603-1
Page MJ, McKenzie JE, Bossuyt PM, et al. (2021) The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 372:n71. Published 2021 Mar 29. https://doi.org/10.1136/bmj.n71
Sterne JA, Hernán MA, Reeves BC, et al. (2016) ROBINS-I: a tool for assessing risk of bias in non-randomised studies of interventions. BMJ. 355:i4919. Published 2016 Oct 12. https://doi.org/10.1136/bmj.i4919
Jensterle M, Jazbinsek S, Bosnjak R, et al. (2019) Advances in the management of craniopharyngioma in children and adults. Radiol Oncol. 53(4):388–396. Published 2019 Oct 25. https://doi.org/10.2478/raon-2019-0036
Kanesaka N, Mikami R, Nakayama H et al (2012) Preliminary results of fractionated stereotactic radiotherapy after cyst drainage for craniopharyngioma in adults. Int J Radiat Oncol Biol Phys 82(4):1356–1360. https://doi.org/10.1016/j.ijrobp.2011.05.014
Schulz-Ertner D, Frank C, Herfarth KK, Rhein B, Wannenmacher M, Debus J (2002) Fractionated stereotactic radiotherapy for craniopharyngiomas. Int J Radiat Oncol Biol Phys 54(4):1114–1120. https://doi.org/10.1016/s0360-3016(02)03029-8
Lopez RC, Martín IT, Ávila RDM, Martínez AMR, Pavón SR (2020) PO-0872: craniopharyngiomas treated with fractionated stereotactic radiotherapy (FSR). Radiother Oncol 152:S468. https://doi.org/10.1016/s0167-8140(21)00889-6
Mizowaki T, Sakanaka K, Ogura K et al (2012) EP-1041 outcomes of fractionated stereotactic radiotherapy in patients with residual or recurrent craniopharyngioma. Radiother Oncol 103:S407–S407. https://doi.org/10.1016/s0167-8140(12)71374-9
Pica A, Abbeel S, Von der Weid N et al (2013) Fractionated stereotactic radiotherapy with static field conformal and non coplanar arcs for pediatric patients with craniopharyngioma: analysis of long term visual outcome and endocrine toxicity. J Radiosurg SBRT 2(3):209–216
Sanz Á, Rodríguez G, Molerón R, Magallón R, García-Berrocal MI (2011) 8724 POSTER fractionated stereotactic radiotherapy (FSRT) for craniopharyngiomas – clinical and radiological control. Eur J Cancer 47:S582–S582. https://doi.org/10.1016/s0959-8049(11)72275-4
Takano S, Akutsu H, Mizumoto M, Yamamoto T, Tsuboi K, Matsumura A (2015) Neuroendoscopy followed by radiotherapy in cystic craniopharyngiomas–a long-term follow-up. World Neurosurg 84(5):1305–15.e152. https://doi.org/10.1016/j.wneu.2015.06.022
Forbes JA, Ordóñez-Rubiano EG, Tomasiewicz HC, et al. (2018) Endonasal endoscopic transsphenoidal resection of intrinsic third ventricular craniopharyngioma: surgical results. J Neurosurg. Published online November 1, 2018. https://doi.org/10.3171/2018.5.JNS18198
Hidalgo ET, Orillac C, Kvint S et al (2020) Quality of life, hypothalamic obesity, and sexual function in adulthood two decades after primary gross-total resection for childhood craniopharyngioma. Childs Nerv Syst 36(2):281–289. https://doi.org/10.1007/s00381-019-04161-9
Madsen PJ, Buch VP, Douglas JE, et al. (2019) Endoscopic endonasal resection versus open surgery for pediatric craniopharyngioma: comparison of outcomes and complications. J Neurosurg Pediatr. Published online June 7, 2019. https://doi.org/10.3171/2019.4.PEDS18612
d’Avella E, Solari D, Somma T et al (2019) The endoscopic endonasal approach for pediatric craniopharyngiomas: the key lessons learned. Childs Nerv Syst 35(11):2147–2155. https://doi.org/10.1007/s00381-019-04168-2
Apra C, Enachescu C, Lapras V, Raverot G, Jouanneau E (2019) Is gross Total resection reasonable in adults with craniopharyngiomas with hypothalamic involvement? World Neurosurg 129:e803–e811. https://doi.org/10.1016/j.wneu.2019.06.037
Grewal MR, Spielman DB, Safi C et al (2020) Gross total versus subtotal surgical resection in the management of craniopharyngiomas. Allergy Rhinol (Providence) 11:2152656720964158. https://doi.org/10.1177/2152656720964158
Gautier A, Godbout A, Grosheny C et al (2012) Markers of recurrence and long-term morbidity in craniopharyngioma: a systematic analysis of 171 patients. J Clin Endocrinol Metab 97(4):1258–1267. https://doi.org/10.1210/jc.2011-2817
Bogusz A, Müller HL (2018) Childhood-onset craniopharyngioma: latest insights into pathology, diagnostics, treatment, and follow-up. Expert Rev Neurother 18(10):793–806. https://doi.org/10.1080/14737175.2018.1528874
Özyurt J, Thiel CM, Lorenzen A et al (2014) Neuropsychological outcome in patients with childhood craniopharyngioma and hypothalamic involvement. J Pediatr 164(4):876-881.e4. https://doi.org/10.1016/j.jpeds.2013.12.010
Aldave G, Okcu MF, Chintagumpala M, et al. (2023) Comparison of neurocognitive and quality-of-life outcomes in pediatric craniopharyngioma patients treated with partial resection and radiotherapy versus gross-total resection only. J Neurosurg Pediatr 31(5):453–462. Published 2023 Feb 17. https://doi.org/10.3171/2022.12.PEDS22367
Willard VW, Berlin KS, Conklin HM, Merchant TE (2019) Trajectories of psychosocial and cognitive functioning in pediatric patients with brain tumors treated with radiation therapy. Neuro Oncol 21(5):678–685. https://doi.org/10.1093/neuonc/noz010
Toussaint L, Indelicato DJ, Muren LP et al (2021) Response to: comments on temporal lobe sparing radiotherapy with photons or protons for cognitive function preservation in paediatric craniopharyngioma by Toussaint, et al. Prior similar field arrangement work and a need for variable RBE Use. Radiother Oncol 158:330–331. https://doi.org/10.1016/j.radonc.2021.01.025
Prieto R, Pascual JM, Subhi-Issa I, Jorquera M, Yus M, Martínez R (2013) Predictive factors for craniopharyngioma recurrence: a systematic review and illustrative case report of a rapid recurrence. World Neurosurg 79(5–6):733–749. https://doi.org/10.1016/j.wneu.2012.07.033
Fischer EG, Welch K, Shillito J Jr, Winston KR, Tarbell NJ (1990) Craniopharyngiomas in children. Long-term effects of conservative surgical procedures combined with radiation therapy. J Neurosurg 73(4):534–540. https://doi.org/10.3171/jns.1990.73.4.0534
Mortini P, Losa M, Pozzobon G et al (2011) Neurosurgical treatment of craniopharyngioma in adults and children: early and long-term results in a large case series. J Neurosurg 114(5):1350–1359. https://doi.org/10.3171/2010.11.JNS10670
Maniker AH, Krieger AJ (1996) Rapid recurrence of craniopharyngioma during pregnancy with recovery of vision: a case report. Surg Neurol 45(4):324–327. https://doi.org/10.1016/0090-3019(95)00399-1
Anegawa S, Hayashi T, Nakagawa S, Furukawa Y, Tomokiyo M (2001) No Shinkei Geka 29(8):727–733
Niu DM, Guo WY, Pan HC, Wong TT (2002) Rapid enlargement of a residual craniopharyngioma during short-term growth hormone replacement. Childs Nerv Syst 18(3–4):164–165. https://doi.org/10.1007/s00381-001-0552-1
Taguchi T, Takao T, Iwasaki Y et al (2010) Rapid recurrence of craniopharyngioma following recombinant human growth hormone replacement. J Neurooncol 100(2):321–322. https://doi.org/10.1007/s11060-010-0161-5
Aydin Y, Can SM, Gŭlkilik A, Tŭrkmenoglu O, Alatli C, Ziyal I (1999) Rapid enlargement and recurrence of a preexisting intrasellar craniopharyngioma during the course of two pregnancies. Case report J Neurosurg 91(2):322–324. https://doi.org/10.3171/jns.1999.91.2.0322
Brastianos PK, Twohy E, Geyer S et al (2023) BRAF-MEK inhibition in newly diagnosed papillary craniopharyngiomas. N Engl J Med 389(2):118–126. https://doi.org/10.1056/NEJMoa2213329
Lehrich BM, Goshtasbi K, Hsu FPK, Kuan EC (2021) Characteristics and overall survival in pediatric versus adult craniopharyngioma: a population-based study. Childs Nerv Syst 37(5):1535–1545. https://doi.org/10.1007/s00381-021-05094-y
Kortmann RD (2011) Different approaches in radiation therapy of craniopharyngioma. Front Endocrinol (Lausanne) 2:100. Published 2011 Dec 20. https://doi.org/10.3389/fendo.2011.00100
Erfurth EM, Holmer H, Fjalldal SB (2013) Mortality and morbidity in adult craniopharyngioma. Pituitary 16(1):46–55. https://doi.org/10.1007/s11102-012-0428-2
O'steen L, Indelicato DJ (2018) Advances in the management of craniopharyngioma. F1000Res 7:F1000 Faculty Rev-1632. Published 2018 Oct 11. https://doi.org/10.12688/f1000research.15834.1
Funding
This research received no specific grant from public, commercial, or not-for-profit funding agencies.
Author information
Authors and Affiliations
Contributions
The project credits are as follows: Lucca B Palavani contributed to conceptualization, methodology, software development, original draft writing, investigation, and data curation. Guilherme Melo Silva was involved in original draft writing, formal analysis, investigation, data curation, and methodology. Pedro Borges contributed to data curation, investigation, original draft writing, and formal analysis. Márcio Yuri Ferreira participated in original draft writing, data curation, and investigation. Marcelo Porto Sousa contributed to original draft writing, data curation, and software development. Marianna G. H. S. J. Leite was involved in original draft writing, data curation, software development, and methodology. Leonardo de Barros Oliveira contributed to writing review and editing, as well as methodology. Sávio Batista was involved in writing review and editing, software development, and formal analysis. Raphael Bertani supervised the project, handled project administration, and participated in writing review and editing. Allan Dias Polverini also supervised the project, handled project administration, and contributed to writing review and editing, as well as visualization. André Beer-Furlan supervised the project and participated in project administration and writing review and editing. Wellingson Paiva contributed to conceptualization, supervision, project administration, writing review and editing, and visualization.
Corresponding author
Ethics declarations
Ethical approval
Not applicable.
Competing interests
The authors declare no competing interests.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.
About this article
Cite this article
Palavani, L.B., Silva, G.M., Borges, P.G.L.B. et al. Fractionated stereotactic radiotherapy in craniopharyngiomas: A systematic review and single arm meta-analysis. J Neurooncol (2024). https://doi.org/10.1007/s11060-024-04621-6
Received:
Accepted:
Published:
DOI: https://doi.org/10.1007/s11060-024-04621-6