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000124245 0247_ $$2doi$$a10.1093/neuonc/now234
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000124245 1001_ $$aMynarek, Martin$$b0
000124245 245__ $$aEvaluation of age-dependent treatment strategies for children and young adults with pineoblastoma: analysis of pooled European Society for Paediatric Oncology (SIOP-E) and US Head Start data.
000124245 260__ $$aOxford$$bOxford Univ. Press$$c2017
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000124245 520__ $$aPineoblastoma is a rare pineal region brain tumor. Treatment strategies have reflected those for other malignant embryonal brain tumors.Original prospective treatment and outcome data from international trial groups were pooled. Cox regression models were developed considering treatment elements as time-dependent covariates.Data on 135 patients with pineoblastoma aged 0.01-20.7 (median 4.9) years were analyzed. Median observation time was 7.3 years. Favorable prognostic factors were age ≥4 years (hazard ratio [HR] for progression-free survival [PFS] 0.270, P < .001) and administration of radiotherapy (HR for PFS 0.282, P < .001). Metastatic disease (HR for PFS 2.015, P = .006), but not postoperative residual tumor, was associated with unfavorable prognosis. In 57 patients <4 years old, 5-year PFS/overall survival (OS) were 11 ± 4%/12 ± 4%. Two patients survived after chemotherapy only, while 3 of 16 treated with craniospinal irradiation (CSI) with boost, and 3 of 5 treated with high-dose chemotherapy (HDCT) and local radiotherapy survived. In 78 patients aged ≥4 years, PFS/OS were 72 ± 7%/73 ± 7% for patients without metastases, and 50 ± 10%/55 ± 10% with metastases. Seventy-three patients received radiotherapy (48 conventionally fractionated CSI, median dose 35.0 [18.0-45.0] Gy, 19 hyperfractionated CSI, 6 local radiotherapy), with (n = 68) or without (n = 6) chemotherapy. The treatment sequence had no impact; application of HDCT had weak impact on survival in older patients.Survival is poor in young children treated without radiotherapy. In these patients, combination of HDCT and local radiotherapy may warrant further evaluation in the absence of more specific or targeted treatments. CSI combined with chemotherapy is effective for older non-metastatic patients.
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000124245 7001_ $$aPizer, Barry$$b1
000124245 7001_ $$aDufour, Christelle$$b2
000124245 7001_ $$avan Vuurden, Dannis$$b3
000124245 7001_ $$aGarami, Miklos$$b4
000124245 7001_ $$aMassimino, Maura$$b5
000124245 7001_ $$aFangusaro, Jason$$b6
000124245 7001_ $$aDavidson, Tom$$b7
000124245 7001_ $$aGil-da-Costa, Maria Joao$$b8
000124245 7001_ $$aSterba, Jaroslav$$b9
000124245 7001_ $$aBenesch, Martin$$b10
000124245 7001_ $$aGerber, Nicolas$$b11
000124245 7001_ $$aJuhnke, B Ole$$b12
000124245 7001_ $$aKwiecien, Robert$$b13
000124245 7001_ $$aPietsch, Torsten$$b14
000124245 7001_ $$0P:(DE-He78)4c28e2aade5f44d8eca9dd8e97638ec8$$aKool, Marcel$$b15$$udkfz
000124245 7001_ $$aClifford, Steve$$b16
000124245 7001_ $$aEllison, David W$$b17
000124245 7001_ $$aGiangaspero, Felice$$b18
000124245 7001_ $$aWesseling, Pieter$$b19
000124245 7001_ $$aGilles, Floyd$$b20
000124245 7001_ $$aGottardo, Nicholas$$b21
000124245 7001_ $$aFinlay, Jonathan L$$b22
000124245 7001_ $$aRutkowski, Stefan$$b23
000124245 7001_ $$avon Hoff, Katja$$b24
000124245 773__ $$0PERI:(DE-600)2094060-9$$a10.1093/neuonc/now234$$gVol. 19, no. 4, p. now234 -$$n4$$p576-585$$tNeuro-Oncology$$v19$$x1523-5866$$y2017
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