Evaluation of age-dependent treatment strategies for children
and young adults with pineoblastoma: analysis of pooled
European Society for Paediatric Oncology (SIOP-E) and US Head
Start data

J 2017

Evaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data

MYNAREK, Martin, Barry PIZER, Christelle DUFOUR, Dannis van VUURDEN, Miklos GARAMI et. al.

Basic information

Original name

Evaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data

Authors

Edition

Neuro-Oncology, Cary, Oxford University Press, 2017, 1522-8517

Other information

Language

English

Type of outcome

Článek v odborném periodiku

Field of Study

30200 3.2 Clinical medicine

Country of publisher

United States of America

Confidentiality degree

není předmětem státního či obchodního tajemství

References:

URL

Impact factor

Impact factor: 9.384

Organization unit

Faculty of Medicine

DOI

http://dx.doi.org/10.1093/neuonc/now234

UT WoS

000400894900015

Keywords in English

high-dose chemotherapy; pediatric; pineoblastoma; radiotherapy; treatment

Abstract

V originále

Background. Pineoblastoma is a rare pineal region brain tumor. Treatment strategies have reflected those for other malignant embryonal brain tumors. Patients and Methods. Original prospective treatment and outcome data from international trial groups were pooled. Cox regression models were developed considering treatment elements as time-dependent covariates. Results. Data on 135 patients with pineoblastoma aged 0.01-20.7 (median 4.9) years were analyzed. Median observation time was 7.3 years. Favorable prognostic factors were age >= 4 years (hazard ratio [HR] for progression-free survival [PFS] 0.270, P < .001) and administration of radiotherapy (HR for PFS 0.282, P < .001). Metastatic disease (HR for PFS 2.015, P =.006), but not postoperative residual tumor, was associated with unfavorable prognosis. In 57 patients < 4 years old, 5-year PFS/overall survival (OS) were 11 +/- 4%/12 +/- 4%. Two patients survived after chemotherapy only, while 3 of 16 treated with craniospinal irradiation (CSI) with boost, and 3 of 5 treated with high-dose chemotherapy (HDCT) and local radiotherapy survived. In 78 patients aged >= 4 years, PFS/OS were 72 +/- 7%/73 +/- 7% for patients without metastases, and 50 +/- 10%/55 +/- 10% with metastases. Seventy-three patients received radiotherapy (48 conventionally fractionated CSI, median dose 35.0 [ 18.0-45.0] Gy, 19 hyperfractionated CSI, 6 local radiotherapy), with (n = 68) or without (n = 6) chemotherapy. The treatment sequence had no impact; application of HDCT had weak impact on survival in older patients. Conclusion. Survival is poor in young children treated without radiotherapy. In these patients, combination of HDCT and local radiotherapy may warrant further evaluation in the absence of more specific or targeted treatments. CSI combined with chemotherapy is effective for older non-metastatic patients.

Citovat

MYNAREK, Martin, Barry PIZER, Christelle DUFOUR, Dannis van VUURDEN, Miklos GARAMI, Maura MASSIMINO, Jason FANGUSARO, Tom DAVIDSON, Maria Joao GIL-DA-COSTA, Jaroslav ŠTĚRBA, Martin BENESCH, Nicolas GERBER, B. Ole JUHNKE, Robert KWIECIEN, Torsten PIETSCH, Marcel KOOL, Steve CLIFFORD, David W. ELLISON, Felice GIANGASPERO, Pieter WESSELING, Floyd GILLES, Nicholas GOTTARDO, Jonathan L. FINLAY, Stefan RUTKOWSKI and Katja von HOFF. Evaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data. Neuro-Oncology. Cary: Oxford University Press, 2017, vol. 19, No 4, p. 576-585. ISSN 1522-8517. Available from: https://dx.doi.org/10.1093/neuonc/now234.

@article{1371294,
   author = {Mynarek, Martin and Pizer, Barry and Dufour, Christelle and Vuurden, Dannis van and Garami, Miklos and Massimino, Maura and Fangusaro, Jason and Davidson, Tom and GilanddaandCosta, Maria Joao and Štěrba, Jaroslav and Benesch, Martin and Gerber, Nicolas and Juhnke, B. Ole and Kwiecien, Robert and Pietsch, Torsten and Kool, Marcel and Clifford, Steve and Ellison, David W. and Giangaspero, Felice and Wesseling, Pieter and Gilles, Floyd and Gottardo, Nicholas and Finlay, Jonathan L. and Rutkowski, Stefan and Hoff, Katja von},
   article_location = {Cary},
   article_number = {4},
   doi = {http://dx.doi.org/10.1093/neuonc/now234},
   keywords = {high-dose chemotherapy; pediatric; pineoblastoma; radiotherapy; treatment},
   language = {eng},
   issn = {1522-8517},
   journal = {Neuro-Oncology},
   title = {Evaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data},
   url = {http://dx.doi.org/10.1093/neuonc/now234},
   volume = {19},
   year = {2017}
}

TY  - JOUR
ID  - 1371294
AU  - Mynarek, Martin - Pizer, Barry - Dufour, Christelle - Vuurden, Dannis van - Garami, Miklos - Massimino, Maura - Fangusaro, Jason - Davidson, Tom - Gil-da-Costa, Maria Joao - Štěrba, Jaroslav - Benesch, Martin - Gerber, Nicolas - Juhnke, B. Ole - Kwiecien, Robert - Pietsch, Torsten - Kool, Marcel - Clifford, Steve - Ellison, David W. - Giangaspero, Felice - Wesseling, Pieter - Gilles, Floyd - Gottardo, Nicholas - Finlay, Jonathan L. - Rutkowski, Stefan - Hoff, Katja von
PY  - 2017
TI  - Evaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data
JF  - Neuro-Oncology
VL  - 19
IS  - 4
SP  - 576-585
EP  - 576-585
PB  - Oxford University Press
SN  - 15228517
KW  - high-dose chemotherapy
KW  - pediatric
KW  - pineoblastoma
KW  - radiotherapy
KW  - treatment
UR  - http://dx.doi.org/10.1093/neuonc/now234
L2  - http://dx.doi.org/10.1093/neuonc/now234
N2  - Background. Pineoblastoma is a rare pineal region brain tumor. Treatment strategies have reflected those for other malignant embryonal brain tumors. Patients and Methods. Original prospective treatment and outcome data from international trial groups were pooled. Cox regression models were developed considering treatment elements as time-dependent covariates. Results. Data on 135 patients with pineoblastoma aged 0.01-20.7 (median 4.9) years were analyzed. Median observation time was 7.3 years. Favorable prognostic factors were age >= 4 years (hazard ratio [HR] for progression-free survival [PFS] 0.270, P < .001) and administration of radiotherapy (HR for PFS 0.282, P < .001). Metastatic disease (HR for PFS 2.015, P =.006), but not postoperative residual tumor, was associated with unfavorable prognosis. In 57 patients < 4 years old, 5-year PFS/overall survival (OS) were 11 +/- 4%/12 +/- 4%. Two patients survived after chemotherapy only, while 3 of 16 treated with craniospinal irradiation (CSI) with boost, and 3 of 5 treated with high-dose chemotherapy (HDCT) and local radiotherapy survived. In 78 patients aged >= 4 years, PFS/OS were 72 +/- 7%/73 +/- 7% for patients without metastases, and 50 +/- 10%/55 +/- 10% with metastases. Seventy-three patients received radiotherapy (48 conventionally fractionated CSI, median dose 35.0 [ 18.0-45.0] Gy, 19 hyperfractionated CSI, 6 local radiotherapy), with (n = 68) or without (n = 6) chemotherapy. The treatment sequence had no impact; application of HDCT had weak impact on survival in older patients. Conclusion. Survival is poor in young children treated without radiotherapy. In these patients, combination of HDCT and local radiotherapy may warrant further evaluation in the absence of more specific or targeted treatments. CSI combined with chemotherapy is effective for older non-metastatic patients.
ER  -

MYNAREK, Martin, Barry PIZER, Christelle DUFOUR, Dannis van VUURDEN, Miklos GARAMI, Maura MASSIMINO, Jason FANGUSARO, Tom DAVIDSON, Maria Joao GIL-DA-COSTA, Jaroslav ŠTĚRBA, Martin BENESCH, Nicolas GERBER, B. Ole JUHNKE, Robert KWIECIEN, Torsten PIETSCH, Marcel KOOL, Steve CLIFFORD, David W. ELLISON, Felice GIANGASPERO, Pieter WESSELING, Floyd GILLES, Nicholas GOTTARDO, Jonathan L. FINLAY, Stefan RUTKOWSKI and Katja von HOFF. Evaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data. \textit{Neuro-Oncology}. Cary: Oxford University Press, 2017, vol.~19, No~4, p.~576-585. ISSN~1522-8517. Available from: https://dx.doi.org/10.1093/neuonc/now234.

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