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

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 & 16 others 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

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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.
Original languageEnglish
Pages (from-to)457-465
Number of pages9
JournalJournal of Neuro-Oncology
DOIs
Publication statusPublished - Aug 14 2018

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Ependymoma
Signs and Symptoms
Pediatrics
Recurrence
Survival
Salvage Therapy

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Pediatric intracranial ependymoma: correlating signs and symptoms at recurrence with outcome in the second prospective AIEOP protocol follow-up. / Massimino, Maura; Barretta, Francesco; Modena, Piergiorgio; Giangaspero, Felice; Chiapparini, Luisa; Erbetta, Alessandra; Boschetti, Luna; Antonelli, Manila; Ferroli, Paolo; Bertin, Daniele; Pecori, Emilia; Biassoni, Veronica; Garrè, Maria Luisa; Schiavello, Elisabetta; Sardi, Iacopo; Viscardi, Elisabetta; Scarzello, Giovanni; Mascarin, Maurizio; Quaglietta, Lucia; Cinalli, Giuseppe; Genitori, Lorenzo; Peretta, Paola; Mussano, Anna; Barra, Salvina; Mastronuzzi, Angela; Giussani, Carlo; Marras, Carlo Efisio; Balter, Rita; Bertolini, Patrizia; Tornesello, Assunta; La Spina, Milena; Buttarelli, Francesca Romana; Ruggiero, Antonio; Caldarelli, Massimo; Poggi, Geraldina; Gandola, Lorenza.

In: Journal of Neuro-Oncology, 14.08.2018, 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, pp. 457-465. https://doi.org/10.1007/s11060-018-2974-6
Massimino, Maura ; Barretta, Francesco ; Modena, Piergiorgio ; Giangaspero, Felice ; Chiapparini, Luisa ; Erbetta, Alessandra ; Boschetti, Luna ; Antonelli, Manila ; Ferroli, Paolo ; Bertin, Daniele ; Pecori, Emilia ; Biassoni, Veronica ; Garrè, Maria Luisa ; Schiavello, Elisabetta ; Sardi, Iacopo ; Viscardi, Elisabetta ; Scarzello, Giovanni ; Mascarin, Maurizio ; Quaglietta, Lucia ; Cinalli, Giuseppe ; Genitori, Lorenzo ; Peretta, Paola ; Mussano, Anna ; Barra, Salvina ; Mastronuzzi, Angela ; Giussani, Carlo ; Marras, Carlo Efisio ; Balter, Rita ; Bertolini, Patrizia ; Tornesello, Assunta ; La Spina, Milena ; Buttarelli, Francesca Romana ; Ruggiero, Antonio ; Caldarelli, Massimo ; Poggi, Geraldina ; Gandola, Lorenza. / 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 ; pp. 457-465.
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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.",
author = "Maura Massimino and Francesco Barretta and Piergiorgio Modena and Felice Giangaspero and Luisa Chiapparini and Alessandra Erbetta and Luna Boschetti and Manila Antonelli and Paolo Ferroli and Daniele Bertin and Emilia Pecori and Veronica Biassoni and Garr{\`e}, {Maria Luisa} and Elisabetta Schiavello and Iacopo Sardi and Elisabetta Viscardi and Giovanni Scarzello and Maurizio Mascarin and Lucia Quaglietta and Giuseppe Cinalli and Lorenzo Genitori and Paola Peretta and Anna Mussano and Salvina Barra and Angela Mastronuzzi and Carlo Giussani and Marras, {Carlo Efisio} and Rita Balter and Patrizia Bertolini and Assunta Tornesello and {La Spina}, Milena and Buttarelli, {Francesca Romana} and Antonio Ruggiero and Massimo Caldarelli and Geraldina Poggi and Lorenza Gandola",
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TY - JOUR

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

AU - Massimino, Maura

AU - Barretta, Francesco

AU - Modena, Piergiorgio

AU - Giangaspero, Felice

AU - Chiapparini, Luisa

AU - Erbetta, Alessandra

AU - Boschetti, Luna

AU - Antonelli, Manila

AU - Ferroli, Paolo

AU - Bertin, Daniele

AU - Pecori, Emilia

AU - Biassoni, Veronica

AU - Garrè, Maria Luisa

AU - Schiavello, Elisabetta

AU - Sardi, Iacopo

AU - Viscardi, Elisabetta

AU - Scarzello, Giovanni

AU - Mascarin, Maurizio

AU - Quaglietta, Lucia

AU - Cinalli, Giuseppe

AU - Genitori, Lorenzo

AU - Peretta, Paola

AU - Mussano, Anna

AU - Barra, Salvina

AU - Mastronuzzi, Angela

AU - Giussani, Carlo

AU - Marras, Carlo Efisio

AU - Balter, Rita

AU - Bertolini, Patrizia

AU - Tornesello, Assunta

AU - La Spina, Milena

AU - Buttarelli, Francesca Romana

AU - Ruggiero, Antonio

AU - Caldarelli, Massimo

AU - Poggi, Geraldina

AU - Gandola, Lorenza

PY - 2018/8/14

Y1 - 2018/8/14

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.

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.

U2 - 10.1007/s11060-018-2974-6

DO - 10.1007/s11060-018-2974-6

M3 - Article

SP - 457

EP - 465

JO - Journal of Neuro-Oncology

JF - Journal of Neuro-Oncology

SN - 0167-594X

ER -