Abstract Objective To study the clinical features of children with recurrent medulloblastoma (MB) and treatment regimens. Methods A retrospective analysis was performed on 101 children with recurrent MB who were admitted to the hospital from August 1, 2011 to July 31, 2017. The children were followed up to July 31, 2020. The Kaplan-Meier method was used for survival analysis. The Cox regression model was used for multivariate regression analysis. Results Of the 101 children, 95 underwent remission induction therapy, among whom 51 had response, resulting in a response rate of 54%. The median overall survival (OS) time after recurrence was 13 months, and the 1-, 3-, and 5-year OS rates were 50.5%±5.0%, 19.8%±4.0%, and 10%±3.3% respectively. There was no significant difference in the 5-year OS rate between the children with different ages (< 3 years or 3-18 years), sexes, pathological types, or Change stages, between the children with or without radiotherapy before recurrence or re-irradiation after recurrence, and between the children with different times to recurrence (< 12 months or ≥ 12 months after surgery) (P > 0.05). There were significant differences in the 5-year OS rate between the children with or without reoperation after recurrence and between the children with different recurrence sites (P < 0.05). The children with reoperation after recurrence had a significantly longer survival time than those without reoperation (P=0.007), and the risk of death in children undergoing reoperation after recurrence was 0.389 times (95% confidence interval:0.196-0.774) that in children who did not undergo such reoperation. Conclusions As for the recurrence of MB, although remission induction therapy again can achieve remission, such children still have a short survival time. Only reoperation can significantly prolong survival time, and therefore, early reoperation can be considered to improve the outcome of children with recurrent MB.
SUN Yan-Ling,LIU Jing-Jing,DU Shu-Xu et al. Survival of children with recurrent medulloblastoma undergoing sequential therapy: an analysis of 101 cases[J]. CJCP, 2021, 23(2): 164-168.
SUN Yan-Ling,LIU Jing-Jing,DU Shu-Xu et al. Survival of children with recurrent medulloblastoma undergoing sequential therapy: an analysis of 101 cases[J]. CJCP, 2021, 23(2): 164-168.
Rolland A, Aquilina K. Surgery for recurrent medulloblastoma:a review[J]. Neurochirurgie, 2019. DOI:10.1016/j.neuchi.2019.06.008. Epub ahead of print.
[2]
Aguilera D, Mazewski C, Fangusaro J, et al. Response to bevacizumab, irinotecan, and temozolomide in children with relapsed medulloblastoma:a multi-institutional experience[J]. Childs Nerv Syst, 2013, 29(4):589-596.
[3]
Sabel M, Fleischhack G, Tippelt S, et al. Relapse patterns and outcome after relapse in standard risk medulloblastoma:a report from the HIT-SIOP-PNET4 study[J]. J Neurooncol, 2016, 129(3):515-524.
[4]
Louis DN, Ohgaki H, Wiestler OD, et al. The 2007 WHO classification of tumours of the central nervous system[J]. Acta Neuropathol, 2007, 114(2):97-109.
[5]
Chang CH, Housepian EM, Herbert C Jr. An operative staging system and a megavoltage radiotherapeutic technic for cerebellar medulloblastomas[J]. Radiology, 1969, 93(6):1351-1359.
[6]
Gururangan S, Krauser J, Watral MA, et al. Efficacy of high-dose chemotherapy or standard salvage therapy in patients with recurrent medulloblastoma[J]. Neuro Oncol, 2008, 10(5):745-751.
[7]
Park JE, Kang J, Yoo KH, et al. Efficacy of high-dose chemotherapy and autologous stem cell transplantation in patients with relapsed medulloblastoma:a report on the Korean Society for Pediatric Neuro-Oncology (KSPNO)-S-053 study[J]. J Korean Med Sci, 2010, 25(8):1160-1166.
[8]
Bomgaars LR, Bernstein M, Krailo M, et al. Phase Ⅱ trial of irinotecan in children with refractory solid tumors:a Children's Oncology Group Study[J]. J Clin Oncol, 2007, 25(29):4622-4627.
[9]
Gerber NU, von Hoff K, Resch A, et al. Treatment of children with central nervous system primitive neuroectodermal tumors/pinealoblastomas in the prospective multicentric trial HIT 2000 using hyperfractionated radiation therapy followed by maintenance chemotherapy[J]. Int J Radiat Oncol Biol Phys, 2014, 89(4):863-871.
[10]
Bautista F, Fioravantti V, de Rojas T, et al. Medulloblastoma in children and adolescents:a systematic review of contemporary phase I and Ⅱ clinical trials and biology update[J]. Cancer Med, 2017, 6(11):2606-2624.
[11]
Taylor MD, Northcott PA, Korshunov A, et al. Molecular subgroups of medulloblastoma:the current consensus[J]. Acta Neuropathol, 2012, 123(4):465-472.
[12]
Louis DN, Perry A, Reifenberger G, et al. The 2016 World Health Organization classification of tumors of the central nervous system:a summary[J]. Acta Neuropathol, 2016, 131(6):803-820.
[13]
Ramaswamy V, Remke M, Bouffet E, et al. Risk stratification of childhood medulloblastoma in the molecular era:the current consensus[J]. Acta Neuropathol, 2016, 131(6):821-831.
Bode U, Zimmermann M, Moser O, et al. Treatment of recurrent primitive neuroectodermal tumors (PNET) in children and adolescents with high-dose chemotherapy (HDC) and stem cell support:results of the HITREZ 97 multicentre trial[J]. J Neurooncol, 2014, 120(3):635-642.
Zhao M, Wang XP, Fu XP, et al. Bevacizumab and stereotactic radiosurgery achieved complete response for pediatric recurrent medulloblastoma[J]. J Cancer Res Ther, 2018, 14(Supplement):S789-S792.
[18]
Rao AD, Rashid AS, Chen QY, et al. Reirradiation for recurrent pediatric central nervous system malignancies:a multi-institutional review[J]. Int J Radiat Oncol Biol Phys, 2017, 99(3):634-641.
[19]
Gupta T, Maitre M, Sastri GJ, et al. Outcomes of salvage re-irradiation in recurrent medulloblastoma correlate with age at initial diagnosis, primary risk-stratification, and molecular subgrouping[J]. J Neurooncol, 2019, 144(2):283-291.
[20]
Gajjar A, Stewart CF, Ellison DW, et al. Phase I study of vismodegib in children with recurrent or refractory medulloblastoma:a pediatric brain tumor consortium study[J]. Clin Cancer Res, 2013, 19(22):6305-6312.
[21]
Kieran MW, Chisholm J, Casanova M, et al. Phase I study of oral sonidegib (LDE225) in pediatric brain and solid tumors and a phase Ⅱ study in children and adults with relapsed medulloblastoma[J]. Neuro Oncol, 2017, 19(11):1542-1552.
[22]
Bakhshinyan D, Venugopal C, Adile AA, et al. BMI1 is a therapeutic target in recurrent medulloblastoma[J]. Oncogene, 2019, 38(10):1702-1716.
[23]
Johnston DL, Keene D, Strother D, et al. Survival following tumor recurrence in children with medulloblastoma[J]. J Pediatr Hematol Oncol, 2018, 40(3):e159-e163.
