PURPOSE: The outcome of recurrent ependymoma in children is dismal. Reirradiation has been proposed as an effective modality for ependymoma at relapse. However, the toxicity and outcome benefits of this approach have not been well established. METHODS AND MATERIALS: We conducted a retrospective population-based study of all patients with recurrent ependymoma treated between 1986 and 2010 in our institution. Demographic, treatment, and outcome data were analyzed for the entire cohort. RESULTS: Of 113 patients with intracranial ependymoma, 47 patients relapsed. At the time of relapse, 29 patients were treated with surgical resection and/or chemotherapy, and 18 patients received full-dose (≥ 54 Gy focal and/or craniospinal) reirradiation with or without surgery at recurrence. Reirradiation was tolerated well with no severe acute complications noticed. Three-year overall survival was 7% ± 6% and 81% ± 12% for nonreirradiated and reirradiated patients, respectively (p < 0.0001). Time to second progression after reirradiation was significantly longer than time to first progression. This surprising phenomenon was associated with improved progression-free survival for tumors with evidence of DNA damage (n = 15; p = 0.002). At a mean follow-up of 3.73 years, only 2/18 patients had endocrine dysfunction, and 1 patient required special education support. However, a decline in intellectual function from pre- to postreirradiation assessment was observed. CONCLUSIONS: Reirradiation is an effective treatment that may change the natural history of recurrent ependymoma in children. However, this change may be associated with increased neurocognitive toxicity. Additional follow-up is needed to determine the risk of late recurrence, secondary radiation-induced tumors, and long-term functional outcome of these patients.
PURPOSE: The outcome of recurrent ependymoma in children is dismal. Reirradiation has been proposed as an effective modality for ependymoma at relapse. However, the toxicity and outcome benefits of this approach have not been well established. METHODS AND MATERIALS: We conducted a retrospective population-based study of all patients with recurrent ependymoma treated between 1986 and 2010 in our institution. Demographic, treatment, and outcome data were analyzed for the entire cohort. RESULTS: Of 113 patients with intracranial ependymoma, 47 patients relapsed. At the time of relapse, 29 patients were treated with surgical resection and/or chemotherapy, and 18 patients received full-dose (≥ 54 Gy focal and/or craniospinal) reirradiation with or without surgery at recurrence. Reirradiation was tolerated well with no severe acute complications noticed. Three-year overall survival was 7% ± 6% and 81% ± 12% for nonreirradiated and reirradiated patients, respectively (p < 0.0001). Time to second progression after reirradiation was significantly longer than time to first progression. This surprising phenomenon was associated with improved progression-free survival for tumors with evidence of DNA damage (n = 15; p = 0.002). At a mean follow-up of 3.73 years, only 2/18 patients had endocrine dysfunction, and 1 patient required special education support. However, a decline in intellectual function from pre- to postreirradiation assessment was observed. CONCLUSIONS: Reirradiation is an effective treatment that may change the natural history of recurrent ependymoma in children. However, this change may be associated with increased neurocognitive toxicity. Additional follow-up is needed to determine the risk of late recurrence, secondary radiation-induced tumors, and long-term functional outcome of these patients.
Authors: Mark W Kieran; Liliana Goumnerova; Peter Manley; Susan N Chi; Karen J Marcus; Andrea G Manzanera; Maria Lucia Silva Polanco; Brian W Guzik; Estuardo Aguilar-Cordova; C Marcela Diaz-Montero; Arthur J DiPatri; Tadanori Tomita; Rishi Lulla; Lianne Greenspan; Laura K Aguilar; Stewart Goldman Journal: Neuro Oncol Date: 2019-03-18 Impact factor: 12.300
Authors: John Glod; Gilbert J Rahme; Harpreet Kaur; Eric H Raabe; Eugene I Hwang; Mark A Israel Journal: J Pediatr Hematol Oncol Date: 2016-05 Impact factor: 1.289
Authors: S C Mack; H Witt; R M Piro; L Gu; S Zuyderduyn; A M Stütz; X Wang; M Gallo; L Garzia; K Zayne; X Zhang; V Ramaswamy; N Jäger; D T W Jones; M Sill; T J Pugh; M Ryzhova; K M Wani; D J H Shih; R Head; M Remke; S D Bailey; T Zichner; C C Faria; M Barszczyk; S Stark; H Seker-Cin; S Hutter; P Johann; S Bender; V Hovestadt; T Tzaridis; A M Dubuc; P A Northcott; J Peacock; K C Bertrand; S Agnihotri; F M G Cavalli; I Clarke; K Nethery-Brokx; C L Creasy; S K Verma; J Koster; X Wu; Y Yao; T Milde; P Sin-Chan; J Zuccaro; L Lau; S Pereira; P Castelo-Branco; M Hirst; M A Marra; S S Roberts; D Fults; L Massimi; Y J Cho; T Van Meter; W Grajkowska; B Lach; A E Kulozik; A von Deimling; O Witt; S W Scherer; X Fan; K M Muraszko; M Kool; S L Pomeroy; N Gupta; J Phillips; A Huang; U Tabori; C Hawkins; D Malkin; P N Kongkham; W A Weiss; N Jabado; J T Rutka; E Bouffet; J O Korbel; M Lupien; K D Aldape; G D Bader; R Eils; P Lichter; P B Dirks; S M Pfister; A Korshunov; M D Taylor Journal: Nature Date: 2014-02-19 Impact factor: 49.962
Authors: Derek S Tsang; Louise Murray; Vijay Ramaswamy; Michal Zapotocky; Uri Tabori; Ute Bartels; Annie Huang; Peter B Dirks; Michael D Taylor; Cynthia Hawkins; Eric Bouffet; Normand Laperriere Journal: Neuro Oncol Date: 2019-03-18 Impact factor: 12.300
Authors: Soumen Khatua; Laurence J N Cooper; David I Sandberg; Leena Ketonen; Jason M Johnson; Michael E Rytting; Diane D Liu; Heather Meador; Prashant Trikha; Robin J Nakkula; Gregory K Behbehani; Dristhi Ragoonanan; Sumit Gupta; Aikaterini Kotrotsou; Tagwa Idris; Elizabeth J Shpall; Katy Rezvani; Rivka Colen; Wafik Zaky; Dean A Lee; Vidya Gopalakrishnan Journal: Neuro Oncol Date: 2020-08-17 Impact factor: 12.300