Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up

M. Massimino, F. Barretta, P. Modena, F. Giangaspero, L. Chiapparini, A. Erbetta, L. Boschetti, M. Antonelli, P. Ferroli, D. Bertin, E. Pecori, V. Biassoni, M.L. Garrè, E. Schiavello, I. Sardi, E. Viscardi, G. Scarzello, M. Mascarin, L. Quaglietta, G. CinalliL. Genitori, P. Peretta, A. Mussano, S. Barra, A. Mastronuzzi, C. Giussani, C.E. Marras, R. Balter, P. Bertolini, A. Tornesello, M. La Spina, F.R. Buttarelli, A. Ruggiero, M. Caldarelli, G. Poggi, L. Gandola

Research output: Contribution to journalArticle

Abstract

Purpose: The aims of patients’ radiological surveillance are to: ascertain relapse; apply second-line therapy; accrue patients in phase 1/2 protocols if second-line therapy is not standardized/curative; and assess/treat iatrogenic effects. To lessen the emotional and socioeconomic burdens for patients and families, we ideally need to establish whether scheduled radiological surveillance gives patients a better outcome than waiting for symptoms and signs to appear. Methods: We analyzed a prospective series of 160 newly-diagnosed and treated pediatric/adolescent patients with intracranial ependymoma, comparing patients with recurrent disease identified on scheduled MRI (the RECPT group; 34 cases) with those showing signs/symptoms of recurrent disease (the SYMPPT group; 16 cases). The median follow-up was 67 months. Results: No significant differences emerged between the two groups in terms of gender, age, tumor grade/site, shunting, residual disease, or type of relapse (local, distant, or concomitant). The time to relapse (median 19 months; range 5–104) and the MRI follow-up intervals did not differ between the SYMPPT and RECPT groups. The presence of signs/symptoms was an unfavorable factor for overall survival (OS) after recurrence (5-year OS: 8% vs. 37%, p = 0.001). On multivariable analysis, an adjusted model confirmed a significantly worse OS in the SYMPPT than in the RECPT patients. Conclusions: Symptomatic relapses carried a significantly worse survival for ependymoma patients than recurrences detected by MRI alone. It would therefore be desirable to identify recurrences before symptoms develop. Radiological follow-up should be retained in ependymoma patient surveillance because there is a chance of salvage treatment for relapses found on MRI. © 2018, Springer Science+Business Media, LLC, part of Springer Nature.
Original languageEnglish
Pages (from-to)457-465
Number of pages9
JournalJournal of Neuro-Oncology
Volume140
Issue number2
DOIs
Publication statusPublished - 2018

Fingerprint

Ependymoma
Signs and Symptoms
Pediatrics
Recurrence
Survival
Salvage Therapy

Keywords

  • Childhood ependymoma
  • Follow-up
  • Re-irradiation
  • Relapse
  • Surveillance
  • cyclophosphamide
  • etoposide
  • vincristine
  • adolescent
  • age
  • Article
  • brain cancer
  • brain surgery
  • cancer grading
  • cancer localization
  • cancer radiotherapy
  • cancer recurrence
  • cancer surgery
  • cancer survival
  • child
  • childhood cancer
  • clinical feature
  • clinical outcome
  • clinical protocol
  • clinical trial
  • cohort analysis
  • controlled study
  • ependymoma
  • female
  • follow up
  • gender
  • human
  • human tissue
  • major clinical study
  • male
  • minimal residual disease
  • nuclear magnetic resonance imaging
  • outcome assessment
  • overall survival
  • pediatric intracranial ependymoma
  • prospective study
  • retrospective study
  • survival rate
  • survival time
  • symptom
  • brain tumor
  • mortality
  • preschool child
  • prognosis
  • tumor recurrence
  • Adolescent
  • Brain Neoplasms
  • Child
  • Child, Preschool
  • Clinical Protocols
  • Ependymoma
  • Female
  • Follow-Up Studies
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Neoplasm Recurrence, Local
  • Prognosis
  • Prospective Studies

Cite this

Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up. / Massimino, M.; Barretta, F.; Modena, P.; Giangaspero, F.; Chiapparini, L.; Erbetta, A.; Boschetti, L.; Antonelli, M.; Ferroli, P.; Bertin, D.; Pecori, E.; Biassoni, V.; Garrè, M.L.; Schiavello, E.; Sardi, I.; Viscardi, E.; Scarzello, G.; Mascarin, M.; Quaglietta, L.; Cinalli, G.; Genitori, L.; Peretta, P.; Mussano, A.; Barra, S.; Mastronuzzi, A.; Giussani, C.; Marras, C.E.; Balter, R.; Bertolini, P.; Tornesello, A.; La Spina, M.; Buttarelli, F.R.; Ruggiero, A.; Caldarelli, M.; Poggi, G.; Gandola, L.

In: Journal of Neuro-Oncology, Vol. 140, No. 2, 2018, p. 457-465.

Research output: Contribution to journalArticle

Massimino, M, Barretta, F, Modena, P, Giangaspero, F, Chiapparini, L, Erbetta, A, Boschetti, L, Antonelli, M, Ferroli, P, Bertin, D, Pecori, E, Biassoni, V, Garrè, ML, Schiavello, E, Sardi, I, Viscardi, E, Scarzello, G, Mascarin, M, Quaglietta, L, Cinalli, G, Genitori, L, Peretta, P, Mussano, A, Barra, S, Mastronuzzi, A, Giussani, C, Marras, CE, Balter, R, Bertolini, P, Tornesello, A, La Spina, M, Buttarelli, FR, Ruggiero, A, Caldarelli, M, Poggi, G & Gandola, L 2018, 'Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up', Journal of Neuro-Oncology, vol. 140, no. 2, pp. 457-465. https://doi.org/10.1007/s11060-018-2974-6
Massimino, M. ; Barretta, F. ; Modena, P. ; Giangaspero, F. ; Chiapparini, L. ; Erbetta, A. ; Boschetti, L. ; Antonelli, M. ; Ferroli, P. ; Bertin, D. ; Pecori, E. ; Biassoni, V. ; Garrè, M.L. ; Schiavello, E. ; Sardi, I. ; Viscardi, E. ; Scarzello, G. ; Mascarin, M. ; Quaglietta, L. ; Cinalli, G. ; Genitori, L. ; Peretta, P. ; Mussano, A. ; Barra, S. ; Mastronuzzi, A. ; Giussani, C. ; Marras, C.E. ; Balter, R. ; Bertolini, P. ; Tornesello, A. ; La Spina, M. ; Buttarelli, F.R. ; Ruggiero, A. ; Caldarelli, M. ; Poggi, G. ; Gandola, L. / Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up. In: Journal of Neuro-Oncology. 2018 ; Vol. 140, No. 2. pp. 457-465.
@article{8beff81b9d6747bbb2f2ab94ac727415,
title = "Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up",
abstract = "Purpose: The aims of patients’ radiological surveillance are to: ascertain relapse; apply second-line therapy; accrue patients in phase 1/2 protocols if second-line therapy is not standardized/curative; and assess/treat iatrogenic effects. To lessen the emotional and socioeconomic burdens for patients and families, we ideally need to establish whether scheduled radiological surveillance gives patients a better outcome than waiting for symptoms and signs to appear. Methods: We analyzed a prospective series of 160 newly-diagnosed and treated pediatric/adolescent patients with intracranial ependymoma, comparing patients with recurrent disease identified on scheduled MRI (the RECPT group; 34 cases) with those showing signs/symptoms of recurrent disease (the SYMPPT group; 16 cases). The median follow-up was 67 months. Results: No significant differences emerged between the two groups in terms of gender, age, tumor grade/site, shunting, residual disease, or type of relapse (local, distant, or concomitant). The time to relapse (median 19 months; range 5–104) and the MRI follow-up intervals did not differ between the SYMPPT and RECPT groups. The presence of signs/symptoms was an unfavorable factor for overall survival (OS) after recurrence (5-year OS: 8{\%} vs. 37{\%}, p = 0.001). On multivariable analysis, an adjusted model confirmed a significantly worse OS in the SYMPPT than in the RECPT patients. Conclusions: Symptomatic relapses carried a significantly worse survival for ependymoma patients than recurrences detected by MRI alone. It would therefore be desirable to identify recurrences before symptoms develop. Radiological follow-up should be retained in ependymoma patient surveillance because there is a chance of salvage treatment for relapses found on MRI. {\circledC} 2018, Springer Science+Business Media, LLC, part of Springer Nature.",
keywords = "Childhood ependymoma, Follow-up, Re-irradiation, Relapse, Surveillance, cyclophosphamide, etoposide, vincristine, adolescent, age, Article, brain cancer, brain surgery, cancer grading, cancer localization, cancer radiotherapy, cancer recurrence, cancer surgery, cancer survival, child, childhood cancer, clinical feature, clinical outcome, clinical protocol, clinical trial, cohort analysis, controlled study, ependymoma, female, follow up, gender, human, human tissue, major clinical study, male, minimal residual disease, nuclear magnetic resonance imaging, outcome assessment, overall survival, pediatric intracranial ependymoma, prospective study, retrospective study, survival rate, survival time, symptom, brain tumor, mortality, preschool child, prognosis, tumor recurrence, Adolescent, Brain Neoplasms, Child, Child, Preschool, Clinical Protocols, Ependymoma, Female, Follow-Up Studies, Humans, Magnetic Resonance Imaging, Male, Neoplasm Recurrence, Local, Prognosis, Prospective Studies",
author = "M. Massimino and F. Barretta and P. Modena and F. Giangaspero and L. Chiapparini and A. Erbetta and L. Boschetti and M. Antonelli and P. Ferroli and D. Bertin and E. Pecori and V. Biassoni and M.L. Garr{\`e} and E. Schiavello and I. Sardi and E. Viscardi and G. Scarzello and M. Mascarin and L. Quaglietta and G. Cinalli and L. Genitori and P. Peretta and A. Mussano and S. Barra and A. Mastronuzzi and C. Giussani and C.E. Marras and R. Balter and P. Bertolini and A. Tornesello and {La Spina}, M. and F.R. Buttarelli and A. Ruggiero and M. Caldarelli and G. Poggi and L. Gandola",
note = "Cited By :1 Export Date: 25 January 2019 CODEN: JNODD Correspondence Address: Massimino, M.; Pediatric Unit (MM, LB, VB, ES), Fondazione IRCCS Istituto Nazionale dei Tumori, Via Venezian 1, Italy; email: maura.massimino@istitutotumori.mi.it Chemicals/CAS: cyclophosphamide, 50-18-0; etoposide, 33419-42-0, 433304-61-1; vincristine, 57-22-7 References: Bouffet, E., Doz, F., Demaille, M.C., Tron, P., Roche, H., Plantaz, D., Thyss, A., Gentet, J.C., Improving survival in recurrent medulloblastoma: earlier detection, better treatment or still an impasse? (1998) Br J Cancer, 77, pp. 1321-1326. , PID: 9579840; Shaw, D.W., Geyer, J.R., Berger, M.S., Milstein, J., Lindsley, K.L., Asymptomatic recurrence detection with surveillance scanning in children with medulloblastoma (1997) J Clin Oncol, 15, pp. 1811-1813. , PID: 9164189; Spreafico, F., Gandola, L., Marchian{\`o}, A., Simonetti, F., Poggi, G., Adduci, A., Clerici, C.A., Massimino, M., Brain magnetic resonance imaging after high-dose chemotherapy and radiotherapy for childhood brain tumors (2008) Int J Radiat Oncol Biol Phys, 70, pp. 1011-1019. , PID: 17904307; Steinbok, P., Hentschel, S., Cochrane, D.D., Kestle, J.R., Value of postoperative surveillance imaging in the management of children with some common brain tumors (1996) J Neurosurg, 84, pp. 726-732. , PID: 8622143; Massimino, M., Miceli, R., Giangaspero, F., Boschetti, L., Modena, P., Antonelli, M., Ferroli, P., Gandola, L., Final results of the second prospective AIEOP protocol for pediatric intracranial ependymoma (2016) Neuro Oncol, 18, pp. 1451-1460. , PID: 27194148; Pajtler, K.W., Mack, S.C., Ramaswamy, V., The current consensus on the clinical management of intracranial ependymoma and its distinct molecular variants (2017) Acta Neuropathol, 133, pp. 5-12. , PID: 27858204; Gessi, M., Giagnacovo, M., Modena, P., Role of immunohistochemistry in the identification of supratentorial C11ORF95-RELA fused ependymoma in routine neuropathology (2017) Am J Surg Pathol; Ramaswamy, V., Hielscher, T., Mack, S.C., Therapeutic impact of cytoreductive surgery and irradiation of posterior fossa ependymoma in the molecular era: a retrospective multicohort analysis (2016) J Clin Oncol, 34 (21), pp. 2468-2477. , PID: 27269943; Kaplan, E.L., Meier, P., Non-parametric estimation from incomplete observation (1958) J Am Stat Assoc, 53 (282), pp. 457-481; Schemper, M., Smith, T.L., A note on quantifying follow-up studies of failure time (1996) Control Clin Trials, 17, pp. 343-346. , PID: 8889347; Durrleman, S., Simon, R., Flexible regression models with cubic splines (1989) Stat Med, 8, pp. 551-561. , PID: 2657958; Concato, J., Peduzzi, P., Holford, T.R., Feinstein, A.R., Importance of events per independent variable in proportional hazards analysis. I. Background, goals, and general strategy (1995) J Clin Epidemiol, 48, pp. 1495-1501. , PID: 8543963; Kagan, A.R., Steckel, R.J., Diagnostic imaging in the surveillance of treated children with cancer (1993) Med Pediatr Oncol, 21, pp. 323-326. , PID: 8492746, Review; Minn, A.Y., Pollock, B.H., Garzarella, L., Dahl, G.V., Kun, L.E., Ducore, J.M., Shibata, A., Fisher, P.G., Surveillance neuroimaging to detect relapse in childhood brain tumors: a Pediatric Oncology Group study (2001) J Clin Oncol, 19, pp. 4135-4140. , PID: 11689581; Yal{\cc}in, B., B{\"u}y{\"u}kpamuk{\cc}u, M., Akalan, N., Cila, A., Kutluk, M.T., Aky{\"u}z, C., Value of surveillance imaging in the management of medulloblastoma (2002) Med Pediatr Oncol, 38, pp. 91-97. , PID: 11813172; Good, C.D., Wade, A.M., Hayward, R.D., Phipps, K.P., Michalski, A.J., Harkness, W.F., Chong, W.K., Surveillance neuroimaging in childhood intracranial ependymoma: how effective, how often, and for how long? (2001) J Neurosurg, 94, pp. 27-32. , PID: 11147894; Antony, R., Wong, K.E., Patel, M., Olch, A.J., McComb, G., Krieger, M., Gilles, F., Finlay, J.L., A retrospective analysis of recurrent intracranial ependymoma (2014) Pediatr Blood Cancer, 61, pp. 1195-1201. , PID: 24615997; Torres, C.F., Rebsamen, S., Silber, J.H., Sutton, L.N., Bilaniuk, L.T., Zimmerman, R.A., Goldwein, J.W., Lange, B.J., Surveillance scanning of children with medulloblastoma (1994) N Engl J Med, 330, pp. 892-895. , PID: 8114859; Kramer, E.D., Vezina, L.G., Packer, R.J., Fitz, C.R., Zimmerman, R.A., Cohen, M.D., Staging and surveillance of children with central nervous system neoplasms: recommendations of the Neurology and Tumor Imaging Committees of the Children’s Cancer Group (1994) Pediatr Neurosurg, 20, pp. 254-262. , PID: 8043464; Howell, L., Mensah, A., Brennan, B., Makin, G., Detection of recurrence in childhood solid tumors (2005) Cancer, 103, pp. 1274-1279. , PID: 15674854; Perreault, S., Lober, R.M., Carret, A.S., Zhang, G., Hershon, L., D{\'e}carie, J.C., Vogel, H., Partap, S., Surveillance imaging in children with malignant CNS tumors: low yield of spine MRI (2014) J Neurooncol, 116, pp. 617-623. , PID: 24401959; Vinchon, M., Leblond, P., Noudel, R., Dhellemmes, P., Intracranial ependymomas in childhood: recurrence, reoperation, and outcome (2005) Childs Nerv Syst, 21, pp. 221-226. , PID: 15599561; Bouffet, E., Hawkins, C.E., Ballourah, W., Taylor, M.D., Bartels, U.K., Schoenhoff, N., Tsangaris, E., Tabori, U., Survival benefit for pediatric patients with recurrent ependymoma treated with reirradiation (2012) Int J Radiat Oncol Biol Phys, 83, pp. 1541-1548. , PID: 22245198; Merchant, T.E., Boop, F.A., Kun, L.E., Sanford, R.A., A retrospective study of surgery and reirradiation for recurrent ependymoma (2008) Int J Radiat Oncol Biol Phys, 71, pp. 87-97. , PID: 18406885; Tsang, D.S., Burghen, E., Klimo, P., Jr., Boop, F.A., Ellison, D.W., Merchant, T.E., Outcomes after reirradiation for recurrent pediatric intracranial ependymoma (2018) Int J Radiat Oncol Biol Phys, 100, pp. 507-515. , PID: 29229328; Ramaswamy, V., Remke, M., Bouffet, E., Recurrence patterns across medulloblastoma subgroups: an integrated clinical and molecular analysis (2013) Lancet Oncol, 14, pp. 1200-1207. , PID: 24140199",
year = "2018",
doi = "10.1007/s11060-018-2974-6",
language = "English",
volume = "140",
pages = "457--465",
journal = "Journal of Neuro-Oncology",
issn = "0167-594X",
publisher = "Springer New York LLC",
number = "2",

}

TY - JOUR

T1 - Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up

AU - Massimino, M.

AU - Barretta, F.

AU - Modena, P.

AU - Giangaspero, F.

AU - Chiapparini, L.

AU - Erbetta, A.

AU - Boschetti, L.

AU - Antonelli, M.

AU - Ferroli, P.

AU - Bertin, D.

AU - Pecori, E.

AU - Biassoni, V.

AU - Garrè, M.L.

AU - Schiavello, E.

AU - Sardi, I.

AU - Viscardi, E.

AU - Scarzello, G.

AU - Mascarin, M.

AU - Quaglietta, L.

AU - Cinalli, G.

AU - Genitori, L.

AU - Peretta, P.

AU - Mussano, A.

AU - Barra, S.

AU - Mastronuzzi, A.

AU - Giussani, C.

AU - Marras, C.E.

AU - Balter, R.

AU - Bertolini, P.

AU - Tornesello, A.

AU - La Spina, M.

AU - Buttarelli, F.R.

AU - Ruggiero, A.

AU - Caldarelli, M.

AU - Poggi, G.

AU - Gandola, L.

N1 - Cited By :1 Export Date: 25 January 2019 CODEN: JNODD Correspondence Address: Massimino, M.; Pediatric Unit (MM, LB, VB, ES), Fondazione IRCCS Istituto Nazionale dei Tumori, Via Venezian 1, Italy; email: maura.massimino@istitutotumori.mi.it Chemicals/CAS: cyclophosphamide, 50-18-0; etoposide, 33419-42-0, 433304-61-1; vincristine, 57-22-7 References: Bouffet, E., Doz, F., Demaille, M.C., Tron, P., Roche, H., Plantaz, D., Thyss, A., Gentet, J.C., Improving survival in recurrent medulloblastoma: earlier detection, better treatment or still an impasse? (1998) Br J Cancer, 77, pp. 1321-1326. , PID: 9579840; Shaw, D.W., Geyer, J.R., Berger, M.S., Milstein, J., Lindsley, K.L., Asymptomatic recurrence detection with surveillance scanning in children with medulloblastoma (1997) J Clin Oncol, 15, pp. 1811-1813. , PID: 9164189; Spreafico, F., Gandola, L., Marchianò, A., Simonetti, F., Poggi, G., Adduci, A., Clerici, C.A., Massimino, M., Brain magnetic resonance imaging after high-dose chemotherapy and radiotherapy for childhood brain tumors (2008) Int J Radiat Oncol Biol Phys, 70, pp. 1011-1019. , PID: 17904307; Steinbok, P., Hentschel, S., Cochrane, D.D., Kestle, J.R., Value of postoperative surveillance imaging in the management of children with some common brain tumors (1996) J Neurosurg, 84, pp. 726-732. , PID: 8622143; Massimino, M., Miceli, R., Giangaspero, F., Boschetti, L., Modena, P., Antonelli, M., Ferroli, P., Gandola, L., Final results of the second prospective AIEOP protocol for pediatric intracranial ependymoma (2016) Neuro Oncol, 18, pp. 1451-1460. , PID: 27194148; Pajtler, K.W., Mack, S.C., Ramaswamy, V., The current consensus on the clinical management of intracranial ependymoma and its distinct molecular variants (2017) Acta Neuropathol, 133, pp. 5-12. , PID: 27858204; Gessi, M., Giagnacovo, M., Modena, P., Role of immunohistochemistry in the identification of supratentorial C11ORF95-RELA fused ependymoma in routine neuropathology (2017) Am J Surg Pathol; Ramaswamy, V., Hielscher, T., Mack, S.C., Therapeutic impact of cytoreductive surgery and irradiation of posterior fossa ependymoma in the molecular era: a retrospective multicohort analysis (2016) J Clin Oncol, 34 (21), pp. 2468-2477. , PID: 27269943; Kaplan, E.L., Meier, P., Non-parametric estimation from incomplete observation (1958) J Am Stat Assoc, 53 (282), pp. 457-481; Schemper, M., Smith, T.L., A note on quantifying follow-up studies of failure time (1996) Control Clin Trials, 17, pp. 343-346. , PID: 8889347; Durrleman, S., Simon, R., Flexible regression models with cubic splines (1989) Stat Med, 8, pp. 551-561. , PID: 2657958; Concato, J., Peduzzi, P., Holford, T.R., Feinstein, A.R., Importance of events per independent variable in proportional hazards analysis. I. Background, goals, and general strategy (1995) J Clin Epidemiol, 48, pp. 1495-1501. , PID: 8543963; Kagan, A.R., Steckel, R.J., Diagnostic imaging in the surveillance of treated children with cancer (1993) Med Pediatr Oncol, 21, pp. 323-326. , PID: 8492746, Review; Minn, A.Y., Pollock, B.H., Garzarella, L., Dahl, G.V., Kun, L.E., Ducore, J.M., Shibata, A., Fisher, P.G., Surveillance neuroimaging to detect relapse in childhood brain tumors: a Pediatric Oncology Group study (2001) J Clin Oncol, 19, pp. 4135-4140. , PID: 11689581; Yalçin, B., Büyükpamukçu, M., Akalan, N., Cila, A., Kutluk, M.T., Akyüz, C., Value of surveillance imaging in the management of medulloblastoma (2002) Med Pediatr Oncol, 38, pp. 91-97. , PID: 11813172; Good, C.D., Wade, A.M., Hayward, R.D., Phipps, K.P., Michalski, A.J., Harkness, W.F., Chong, W.K., Surveillance neuroimaging in childhood intracranial ependymoma: how effective, how often, and for how long? (2001) J Neurosurg, 94, pp. 27-32. , PID: 11147894; Antony, R., Wong, K.E., Patel, M., Olch, A.J., McComb, G., Krieger, M., Gilles, F., Finlay, J.L., A retrospective analysis of recurrent intracranial ependymoma (2014) Pediatr Blood Cancer, 61, pp. 1195-1201. , PID: 24615997; Torres, C.F., Rebsamen, S., Silber, J.H., Sutton, L.N., Bilaniuk, L.T., Zimmerman, R.A., Goldwein, J.W., Lange, B.J., Surveillance scanning of children with medulloblastoma (1994) N Engl J Med, 330, pp. 892-895. , PID: 8114859; Kramer, E.D., Vezina, L.G., Packer, R.J., Fitz, C.R., Zimmerman, R.A., Cohen, M.D., Staging and surveillance of children with central nervous system neoplasms: recommendations of the Neurology and Tumor Imaging Committees of the Children’s Cancer Group (1994) Pediatr Neurosurg, 20, pp. 254-262. , PID: 8043464; Howell, L., Mensah, A., Brennan, B., Makin, G., Detection of recurrence in childhood solid tumors (2005) Cancer, 103, pp. 1274-1279. , PID: 15674854; Perreault, S., Lober, R.M., Carret, A.S., Zhang, G., Hershon, L., Décarie, J.C., Vogel, H., Partap, S., Surveillance imaging in children with malignant CNS tumors: low yield of spine MRI (2014) J Neurooncol, 116, pp. 617-623. , PID: 24401959; Vinchon, M., Leblond, P., Noudel, R., Dhellemmes, P., Intracranial ependymomas in childhood: recurrence, reoperation, and outcome (2005) Childs Nerv Syst, 21, pp. 221-226. , PID: 15599561; Bouffet, E., Hawkins, C.E., Ballourah, W., Taylor, M.D., Bartels, U.K., Schoenhoff, N., Tsangaris, E., Tabori, U., Survival benefit for pediatric patients with recurrent ependymoma treated with reirradiation (2012) Int J Radiat Oncol Biol Phys, 83, pp. 1541-1548. , PID: 22245198; Merchant, T.E., Boop, F.A., Kun, L.E., Sanford, R.A., A retrospective study of surgery and reirradiation for recurrent ependymoma (2008) Int J Radiat Oncol Biol Phys, 71, pp. 87-97. , PID: 18406885; Tsang, D.S., Burghen, E., Klimo, P., Jr., Boop, F.A., Ellison, D.W., Merchant, T.E., Outcomes after reirradiation for recurrent pediatric intracranial ependymoma (2018) Int J Radiat Oncol Biol Phys, 100, pp. 507-515. , PID: 29229328; Ramaswamy, V., Remke, M., Bouffet, E., Recurrence patterns across medulloblastoma subgroups: an integrated clinical and molecular analysis (2013) Lancet Oncol, 14, pp. 1200-1207. , PID: 24140199

PY - 2018

Y1 - 2018

N2 - Purpose: The aims of patients’ radiological surveillance are to: ascertain relapse; apply second-line therapy; accrue patients in phase 1/2 protocols if second-line therapy is not standardized/curative; and assess/treat iatrogenic effects. To lessen the emotional and socioeconomic burdens for patients and families, we ideally need to establish whether scheduled radiological surveillance gives patients a better outcome than waiting for symptoms and signs to appear. Methods: We analyzed a prospective series of 160 newly-diagnosed and treated pediatric/adolescent patients with intracranial ependymoma, comparing patients with recurrent disease identified on scheduled MRI (the RECPT group; 34 cases) with those showing signs/symptoms of recurrent disease (the SYMPPT group; 16 cases). The median follow-up was 67 months. Results: No significant differences emerged between the two groups in terms of gender, age, tumor grade/site, shunting, residual disease, or type of relapse (local, distant, or concomitant). The time to relapse (median 19 months; range 5–104) and the MRI follow-up intervals did not differ between the SYMPPT and RECPT groups. The presence of signs/symptoms was an unfavorable factor for overall survival (OS) after recurrence (5-year OS: 8% vs. 37%, p = 0.001). On multivariable analysis, an adjusted model confirmed a significantly worse OS in the SYMPPT than in the RECPT patients. Conclusions: Symptomatic relapses carried a significantly worse survival for ependymoma patients than recurrences detected by MRI alone. It would therefore be desirable to identify recurrences before symptoms develop. Radiological follow-up should be retained in ependymoma patient surveillance because there is a chance of salvage treatment for relapses found on MRI. © 2018, Springer Science+Business Media, LLC, part of Springer Nature.

AB - Purpose: The aims of patients’ radiological surveillance are to: ascertain relapse; apply second-line therapy; accrue patients in phase 1/2 protocols if second-line therapy is not standardized/curative; and assess/treat iatrogenic effects. To lessen the emotional and socioeconomic burdens for patients and families, we ideally need to establish whether scheduled radiological surveillance gives patients a better outcome than waiting for symptoms and signs to appear. Methods: We analyzed a prospective series of 160 newly-diagnosed and treated pediatric/adolescent patients with intracranial ependymoma, comparing patients with recurrent disease identified on scheduled MRI (the RECPT group; 34 cases) with those showing signs/symptoms of recurrent disease (the SYMPPT group; 16 cases). The median follow-up was 67 months. Results: No significant differences emerged between the two groups in terms of gender, age, tumor grade/site, shunting, residual disease, or type of relapse (local, distant, or concomitant). The time to relapse (median 19 months; range 5–104) and the MRI follow-up intervals did not differ between the SYMPPT and RECPT groups. The presence of signs/symptoms was an unfavorable factor for overall survival (OS) after recurrence (5-year OS: 8% vs. 37%, p = 0.001). On multivariable analysis, an adjusted model confirmed a significantly worse OS in the SYMPPT than in the RECPT patients. Conclusions: Symptomatic relapses carried a significantly worse survival for ependymoma patients than recurrences detected by MRI alone. It would therefore be desirable to identify recurrences before symptoms develop. Radiological follow-up should be retained in ependymoma patient surveillance because there is a chance of salvage treatment for relapses found on MRI. © 2018, Springer Science+Business Media, LLC, part of Springer Nature.

KW - Childhood ependymoma

KW - Follow-up

KW - Re-irradiation

KW - Relapse

KW - Surveillance

KW - cyclophosphamide

KW - etoposide

KW - vincristine

KW - adolescent

KW - age

KW - Article

KW - brain cancer

KW - brain surgery

KW - cancer grading

KW - cancer localization

KW - cancer radiotherapy

KW - cancer recurrence

KW - cancer surgery

KW - cancer survival

KW - child

KW - childhood cancer

KW - clinical feature

KW - clinical outcome

KW - clinical protocol

KW - clinical trial

KW - cohort analysis

KW - controlled study

KW - ependymoma

KW - female

KW - follow up

KW - gender

KW - human

KW - human tissue

KW - major clinical study

KW - male

KW - minimal residual disease

KW - nuclear magnetic resonance imaging

KW - outcome assessment

KW - overall survival

KW - pediatric intracranial ependymoma

KW - prospective study

KW - retrospective study

KW - survival rate

KW - survival time

KW - symptom

KW - brain tumor

KW - mortality

KW - preschool child

KW - prognosis

KW - tumor recurrence

KW - Adolescent

KW - Brain Neoplasms

KW - Child

KW - Child, Preschool

KW - Clinical Protocols

KW - Ependymoma

KW - Female

KW - Follow-Up Studies

KW - Humans

KW - Magnetic Resonance Imaging

KW - Male

KW - Neoplasm Recurrence, Local

KW - Prognosis

KW - Prospective Studies

U2 - 10.1007/s11060-018-2974-6

DO - 10.1007/s11060-018-2974-6

M3 - Article

VL - 140

SP - 457

EP - 465

JO - Journal of Neuro-Oncology

JF - Journal of Neuro-Oncology

SN - 0167-594X

IS - 2

ER -