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Erschienen in: Journal of Neuro-Oncology 2/2018

14.08.2018 | Clinical Study

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

verfasst von: Maura Massimino, Francesco Barretta, Piergiorgio Modena, Felice Giangaspero, Luisa Chiapparini, Alessandra Erbetta, Luna Boschetti, Manila Antonelli, Paolo Ferroli, Daniele Bertin, Emilia Pecori, Veronica Biassoni, Maria Luisa Garrè, Elisabetta Schiavello, Iacopo Sardi, Elisabetta Viscardi, Giovanni Scarzello, Maurizio Mascarin, Lucia Quaglietta, Giuseppe Cinalli, Lorenzo Genitori, Paola Peretta, Anna Mussano, Salvina Barra, Angela Mastronuzzi, Carlo Giussani, Carlo Efisio Marras, Rita Balter, Patrizia Bertolini, Assunta Tornesello, Milena La Spina, Francesca Romana Buttarelli, Antonio Ruggiero, Massimo Caldarelli, Geraldina Poggi, Lorenza Gandola

Erschienen in: Journal of Neuro-Oncology | Ausgabe 2/2018

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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.
Literatur
1.
Zurück zum Zitat Bouffet E, Doz F, Demaille MC, Tron P, Roche H, Plantaz D, Thyss A, Stephan JL, Lejars O, Sariban E, Buclon M, Zücker JM, Brunat-Mentigny M, Bernard JL, Gentet JC (1998) Improving survival in recurrent medulloblastoma: earlier detection, better treatment or still an impasse? Br J Cancer 77:1321–1326CrossRefPubMedPubMedCentral Bouffet E, Doz F, Demaille MC, Tron P, Roche H, Plantaz D, Thyss A, Stephan JL, Lejars O, Sariban E, Buclon M, Zücker JM, Brunat-Mentigny M, Bernard JL, Gentet JC (1998) Improving survival in recurrent medulloblastoma: earlier detection, better treatment or still an impasse? Br J Cancer 77:1321–1326CrossRefPubMedPubMedCentral
2.
Zurück zum Zitat Shaw DW, Geyer JR, Berger MS, Milstein J, Lindsley KL (1997) Asymptomatic recurrence detection with surveillance scanning in children with medulloblastoma. J Clin Oncol 15:1811–1813CrossRefPubMed Shaw DW, Geyer JR, Berger MS, Milstein J, Lindsley KL (1997) Asymptomatic recurrence detection with surveillance scanning in children with medulloblastoma. J Clin Oncol 15:1811–1813CrossRefPubMed
3.
Zurück zum Zitat Spreafico F, Gandola L, Marchianò A, Simonetti F, Poggi G, Adduci A, Clerici CA, Luksch R, Biassoni V, Meazza C, Catania S, Terenziani M, Musumeci R, Fossati-Bellani F, Massimino M (2008) Brain magnetic resonance imaging after high-dose chemotherapy and radiotherapy for childhood brain tumors. Int J Radiat Oncol Biol Phys 70:1011–1019CrossRefPubMed Spreafico F, Gandola L, Marchianò A, Simonetti F, Poggi G, Adduci A, Clerici CA, Luksch R, Biassoni V, Meazza C, Catania S, Terenziani M, Musumeci R, Fossati-Bellani F, Massimino M (2008) Brain magnetic resonance imaging after high-dose chemotherapy and radiotherapy for childhood brain tumors. Int J Radiat Oncol Biol Phys 70:1011–1019CrossRefPubMed
4.
Zurück zum Zitat Steinbok P, Hentschel S, Cochrane DD, Kestle JR (1996) Value of postoperative surveillance imaging in the management of children with some common brain tumors. J Neurosurg 84:726–732CrossRefPubMed Steinbok P, Hentschel S, Cochrane DD, Kestle JR (1996) Value of postoperative surveillance imaging in the management of children with some common brain tumors. J Neurosurg 84:726–732CrossRefPubMed
5.
Zurück zum Zitat Massimino M, Miceli R, Giangaspero F, Boschetti L, Modena P, Antonelli M, Ferroli P, Bertin D, Pecori E, Valentini L, Biassoni V, Garrè ML, Schiavello E, Sardi I, Cama A, Viscardi E, Scarzello G, Scoccianti S, Mascarin M, Quaglietta L, Cinalli G, Diletto B, Genitori L, Peretta P, Mussano A, Buccoliero A, Calareso G, Barra S, Mastronuzzi A, Giussani C, Marras CE, Balter R, Bertolini P, Giombelli E, La Spina M, Buttarelli FR, Pollo B, Gandola L (2016) Final results of the second prospective AIEOP protocol for pediatric intracranial ependymoma. Neuro Oncol 18:1451–1460CrossRefPubMedPubMedCentral Massimino M, Miceli R, Giangaspero F, Boschetti L, Modena P, Antonelli M, Ferroli P, Bertin D, Pecori E, Valentini L, Biassoni V, Garrè ML, Schiavello E, Sardi I, Cama A, Viscardi E, Scarzello G, Scoccianti S, Mascarin M, Quaglietta L, Cinalli G, Diletto B, Genitori L, Peretta P, Mussano A, Buccoliero A, Calareso G, Barra S, Mastronuzzi A, Giussani C, Marras CE, Balter R, Bertolini P, Giombelli E, La Spina M, Buttarelli FR, Pollo B, Gandola L (2016) Final results of the second prospective AIEOP protocol for pediatric intracranial ependymoma. Neuro Oncol 18:1451–1460CrossRefPubMedPubMedCentral
6.
Zurück zum Zitat Pajtler KW, Mack SC, Ramaswamy V et al (2017) The current consensus on the clinical management of intracranial ependymoma and its distinct molecular variants. Acta Neuropathol 133:5–12CrossRefPubMed Pajtler KW, Mack SC, Ramaswamy V et al (2017) The current consensus on the clinical management of intracranial ependymoma and its distinct molecular variants. Acta Neuropathol 133:5–12CrossRefPubMed
8.
Zurück zum Zitat Ramaswamy V, Hielscher T, Mack SC et al (2016) Therapeutic impact of cytoreductive surgery and irradiation of posterior fossa ependymoma in the molecular era: a retrospective multicohort analysis. J Clin Oncol 34(21):2468–2477CrossRefPubMedPubMedCentral Ramaswamy V, Hielscher T, Mack SC et al (2016) Therapeutic impact of cytoreductive surgery and irradiation of posterior fossa ependymoma in the molecular era: a retrospective multicohort analysis. J Clin Oncol 34(21):2468–2477CrossRefPubMedPubMedCentral
9.
Zurück zum Zitat Kaplan EL, Meier P (1958) Non-parametric estimation from incomplete observation. J Am Stat Assoc 53(282):457–481CrossRef Kaplan EL, Meier P (1958) Non-parametric estimation from incomplete observation. J Am Stat Assoc 53(282):457–481CrossRef
10.
Zurück zum Zitat Schemper M, Smith TL (1996) A note on quantifying follow-up studies of failure time. Control Clin Trials 17:343–346CrossRefPubMed Schemper M, Smith TL (1996) A note on quantifying follow-up studies of failure time. Control Clin Trials 17:343–346CrossRefPubMed
11.
Zurück zum Zitat Durrleman S, Simon R (1989) Flexible regression models with cubic splines. Stat Med 8:551–561CrossRefPubMed Durrleman S, Simon R (1989) Flexible regression models with cubic splines. Stat Med 8:551–561CrossRefPubMed
12.
Zurück zum Zitat Concato J, Peduzzi P, Holford TR, Feinstein AR (1995) Importance of events per independent variable in proportional hazards analysis. I. Background, goals, and general strategy. J Clin Epidemiol 48:1495–1501CrossRefPubMed Concato J, Peduzzi P, Holford TR, Feinstein AR (1995) Importance of events per independent variable in proportional hazards analysis. I. Background, goals, and general strategy. J Clin Epidemiol 48:1495–1501CrossRefPubMed
13.
Zurück zum Zitat Kagan AR, Steckel RJ (1993) Diagnostic imaging in the surveillance of treated children with cancer. Med Pediatr Oncol 21:323–326. ReviewCrossRefPubMed Kagan AR, Steckel RJ (1993) Diagnostic imaging in the surveillance of treated children with cancer. Med Pediatr Oncol 21:323–326. ReviewCrossRefPubMed
14.
Zurück zum Zitat Minn AY, Pollock BH, Garzarella L, Dahl GV, Kun LE, Ducore JM, Shibata A, Kepner J, Fisher PG (2001) Surveillance neuroimaging to detect relapse in childhood brain tumors: a Pediatric Oncology Group study. J Clin Oncol 19:4135–4140CrossRefPubMed Minn AY, Pollock BH, Garzarella L, Dahl GV, Kun LE, Ducore JM, Shibata A, Kepner J, Fisher PG (2001) Surveillance neuroimaging to detect relapse in childhood brain tumors: a Pediatric Oncology Group study. J Clin Oncol 19:4135–4140CrossRefPubMed
15.
Zurück zum Zitat Yalçin B, Büyükpamukçu M, Akalan N, Cila A, Kutluk MT, Akyüz C (2002) Value of surveillance imaging in the management of medulloblastoma. Med Pediatr Oncol 38:91–97CrossRefPubMed Yalçin B, Büyükpamukçu M, Akalan N, Cila A, Kutluk MT, Akyüz C (2002) Value of surveillance imaging in the management of medulloblastoma. Med Pediatr Oncol 38:91–97CrossRefPubMed
16.
Zurück zum Zitat Good CD, Wade AM, Hayward RD, Phipps KP, Michalski AJ, Harkness WF, Chong WK (2001) Surveillance neuroimaging in childhood intracranial ependymoma: how effective, how often, and for how long? J Neurosurg 94:27–32CrossRefPubMed Good CD, Wade AM, Hayward RD, Phipps KP, Michalski AJ, Harkness WF, Chong WK (2001) Surveillance neuroimaging in childhood intracranial ependymoma: how effective, how often, and for how long? J Neurosurg 94:27–32CrossRefPubMed
17.
Zurück zum Zitat Antony R, Wong KE, Patel M, Olch AJ, McComb G, Krieger M, Gilles F, Sposto R, Erdreich-Epstein A, Dhall G, Gardner S, Finlay JL (2014) A retrospective analysis of recurrent intracranial ependymoma. Pediatr Blood Cancer 61:1195–1201CrossRefPubMed Antony R, Wong KE, Patel M, Olch AJ, McComb G, Krieger M, Gilles F, Sposto R, Erdreich-Epstein A, Dhall G, Gardner S, Finlay JL (2014) A retrospective analysis of recurrent intracranial ependymoma. Pediatr Blood Cancer 61:1195–1201CrossRefPubMed
18.
Zurück zum Zitat Torres CF, Rebsamen S, Silber JH, Sutton LN, Bilaniuk LT, Zimmerman RA, Goldwein JW, Phillips PC, Lange BJ (1994) Surveillance scanning of children with medulloblastoma. N Engl J Med 330:892–895CrossRefPubMed Torres CF, Rebsamen S, Silber JH, Sutton LN, Bilaniuk LT, Zimmerman RA, Goldwein JW, Phillips PC, Lange BJ (1994) Surveillance scanning of children with medulloblastoma. N Engl J Med 330:892–895CrossRefPubMed
19.
Zurück zum Zitat Kramer ED, Vezina LG, Packer RJ, Fitz CR, Zimmerman RA, Cohen MD (1994) Staging and surveillance of children with central nervous system neoplasms: recommendations of the Neurology and Tumor Imaging Committees of the Children’s Cancer Group. Pediatr Neurosurg 20:254–262CrossRefPubMed Kramer ED, Vezina LG, Packer RJ, Fitz CR, Zimmerman RA, Cohen MD (1994) Staging and surveillance of children with central nervous system neoplasms: recommendations of the Neurology and Tumor Imaging Committees of the Children’s Cancer Group. Pediatr Neurosurg 20:254–262CrossRefPubMed
20.
Zurück zum Zitat Howell L, Mensah A, Brennan B, Makin G (2005) Detection of recurrence in childhood solid tumors. Cancer 103:1274–1279CrossRefPubMed Howell L, Mensah A, Brennan B, Makin G (2005) Detection of recurrence in childhood solid tumors. Cancer 103:1274–1279CrossRefPubMed
21.
Zurück zum Zitat Perreault S, Lober RM, Carret AS, Zhang G, Hershon L, Décarie JC, Vogel H, Yeom KW, Fisher PG, Partap S (2014) Surveillance imaging in children with malignant CNS tumors: low yield of spine MRI. J Neurooncol 116:617–623CrossRefPubMed Perreault S, Lober RM, Carret AS, Zhang G, Hershon L, Décarie JC, Vogel H, Yeom KW, Fisher PG, Partap S (2014) Surveillance imaging in children with malignant CNS tumors: low yield of spine MRI. J Neurooncol 116:617–623CrossRefPubMed
22.
Zurück zum Zitat Vinchon M, Leblond P, Noudel R, Dhellemmes P (2005) Intracranial ependymomas in childhood: recurrence, reoperation, and outcome. Childs Nerv Syst 21:221–226CrossRefPubMed Vinchon M, Leblond P, Noudel R, Dhellemmes P (2005) Intracranial ependymomas in childhood: recurrence, reoperation, and outcome. Childs Nerv Syst 21:221–226CrossRefPubMed
23.
Zurück zum Zitat Bouffet E, Hawkins CE, Ballourah W, Taylor MD, Bartels UK, Schoenhoff N, Tsangaris E, Huang A, Kulkarni A, Mabbot DJ, Laperriere N, Tabori U (2012) Survival benefit for pediatric patients with recurrent ependymoma treated with reirradiation. Int J Radiat Oncol Biol Phys 83:1541–1548CrossRefPubMed Bouffet E, Hawkins CE, Ballourah W, Taylor MD, Bartels UK, Schoenhoff N, Tsangaris E, Huang A, Kulkarni A, Mabbot DJ, Laperriere N, Tabori U (2012) Survival benefit for pediatric patients with recurrent ependymoma treated with reirradiation. Int J Radiat Oncol Biol Phys 83:1541–1548CrossRefPubMed
24.
Zurück zum Zitat Merchant TE, Boop FA, Kun LE, Sanford RA (2008) A retrospective study of surgery and reirradiation for recurrent ependymoma. Int J Radiat Oncol Biol Phys 71:87–97CrossRefPubMed Merchant TE, Boop FA, Kun LE, Sanford RA (2008) A retrospective study of surgery and reirradiation for recurrent ependymoma. Int J Radiat Oncol Biol Phys 71:87–97CrossRefPubMed
25.
Zurück zum Zitat Tsang DS, Burghen E, Klimo P Jr, Boop FA, Ellison DW, Merchant TE (2018) Outcomes after reirradiation for recurrent pediatric intracranial ependymoma. Int J Radiat Oncol Biol Phys 100:507–515CrossRefPubMed Tsang DS, Burghen E, Klimo P Jr, Boop FA, Ellison DW, Merchant TE (2018) Outcomes after reirradiation for recurrent pediatric intracranial ependymoma. Int J Radiat Oncol Biol Phys 100:507–515CrossRefPubMed
26.
Zurück zum Zitat Ramaswamy V, Remke M, Bouffet E et al (2013) Recurrence patterns across medulloblastoma subgroups: an integrated clinical and molecular analysis. Lancet Oncol 14:1200–1207CrossRefPubMedPubMedCentral Ramaswamy V, Remke M, Bouffet E et al (2013) Recurrence patterns across medulloblastoma subgroups: an integrated clinical and molecular analysis. Lancet Oncol 14:1200–1207CrossRefPubMedPubMedCentral
Metadaten
Titel
Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up
verfasst von
Maura Massimino
Francesco Barretta
Piergiorgio Modena
Felice Giangaspero
Luisa Chiapparini
Alessandra Erbetta
Luna Boschetti
Manila Antonelli
Paolo Ferroli
Daniele Bertin
Emilia Pecori
Veronica Biassoni
Maria Luisa Garrè
Elisabetta Schiavello
Iacopo Sardi
Elisabetta Viscardi
Giovanni Scarzello
Maurizio Mascarin
Lucia Quaglietta
Giuseppe Cinalli
Lorenzo Genitori
Paola Peretta
Anna Mussano
Salvina Barra
Angela Mastronuzzi
Carlo Giussani
Carlo Efisio Marras
Rita Balter
Patrizia Bertolini
Assunta Tornesello
Milena La Spina
Francesca Romana Buttarelli
Antonio Ruggiero
Massimo Caldarelli
Geraldina Poggi
Lorenza Gandola
Publikationsdatum
14.08.2018
Verlag
Springer US
Erschienen in
Journal of Neuro-Oncology / Ausgabe 2/2018
Print ISSN: 0167-594X
Elektronische ISSN: 1573-7373
DOI
https://doi.org/10.1007/s11060-018-2974-6

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