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000168085 1001_ $$00000-0001-6101-149X$$aUpadhyaya, Santhosh A$$b0
000168085 245__ $$aRelevance of Molecular Groups in Children with Newly Diagnosed Atypical Teratoid Rhabdoid Tumor: Results from Prospective St. Jude Multi-Institutional Trials.
000168085 260__ $$aPhiladelphia, Pa. [u.a.]$$bAACR$$c2021
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000168085 520__ $$aReport relevance of molecular groups to clinico-pathologic features, germline SMARCB1/SMARCA4 alterations (GLA), and survival of children with atypical teratoid rhabdoid tumor (ATRT) treated in two multi-institutional clinical trials.Seventy-four participants with newly diagnosed ATRT were treated in two trials: infants (SJYC07: age<3 years; n=52) and children (SJMB03: age 3-21 years; n=22), using surgery, conventional chemotherapy (infants), or dose-dense chemotherapy with autologous stem cell rescue (children), and age- and risk-adapted radiation therapy [focal (infants) and craniospinal (CSI) (children)]. Molecular groups ATRT-MYC (MYC), ATRT-SHH (SHH), and ATRT-TYR (TYR) were determined from tumor DNA methylation profiles.Twenty-four participants (32%) were alive at time of analysis at a median follow-up of 8.4 years (range, 3.1-14.1 years). Methylation profiling classified 64 ATRTs as TYR (n=21), SHH (n=30), and MYC (n=13), SHH group being associated with metastatic disease. Among infants, TYR group had the best overall survival (OS) (P=0.02). However, outcomes did not differ by molecular groups among infants with non-metastatic (M0) disease. Children with M0 disease and <1.5 cm2 residual tumor had a 5-year progression-free survival (PFS) of 72.7{plus minus}12.7% and OS of 81.8{plus minus}11%. Infants with M0 disease had a 5-year PFS of 39.1{plus minus}11.5% and OS of 51.8{plus minus}12%. Those with metastases fared poorly [5-year OS 25{plus minus}12.5% (children) and 0% (infants)]. SMARCB1 GLAs were not associated with PFS.Among infants, those with ATRT-TYR had the best OS. ATRT-SHH was associated with metastases and consequently with inferior outcomes. Children with non-metastatic ATRT benefit from post-operative CSI and adjuvant chemotherapy.
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000168085 7001_ $$00000-0001-7441-9486$$aRobinson, Giles$$b1
000168085 7001_ $$aOnar-Thomas, Arzu$$b2
000168085 7001_ $$00000-0003-0997-4728$$aOrr, Brent A$$b3
000168085 7001_ $$aJohann, Pascal$$b4
000168085 7001_ $$aWu, Gang$$b5
000168085 7001_ $$aBillups, Catherine A$$b6
000168085 7001_ $$00000-0002-1653-3026$$aTatevossian, Ruth G$$b7
000168085 7001_ $$00000-0003-1381-7434$$aDhanda, Sandeep Kumar$$b8
000168085 7001_ $$aSrinivasan, Ashok$$b9
000168085 7001_ $$aBroniscer, Alberto$$b10
000168085 7001_ $$aQaddoumi, Ibrahim$$b11
000168085 7001_ $$aVinitsky, Anna$$b12
000168085 7001_ $$aArmstrong, Gregory T$$b13
000168085 7001_ $$00000-0002-2062-0974$$aBendel, Anne$$b14
000168085 7001_ $$00000-0002-7883-3710$$aHassall, Timothy Eg$$b15
000168085 7001_ $$00000-0001-5615-7689$$aPartap, Sonia$$b16
000168085 7001_ $$aFisher, Paul G$$b17
000168085 7001_ $$00000-0002-4100-7001$$aCrawford, John R$$b18
000168085 7001_ $$aChintagumpala, Murali M$$b19
000168085 7001_ $$00000-0002-6832-6539$$aBouffet, Eric$$b20
000168085 7001_ $$aGururangan, Sridharan$$b21
000168085 7001_ $$aMostafavi, Roya$$b22
000168085 7001_ $$aSanders, Robert P$$b23
000168085 7001_ $$aKlimo, Paul$$b24
000168085 7001_ $$00000-0003-1479-9864$$aPatay, Zoltan$$b25
000168085 7001_ $$aIndelicato, Daniel J$$b26
000168085 7001_ $$00000-0002-5581-6555$$aNichols, Kim E$$b27
000168085 7001_ $$aBoop, Frederick A$$b28
000168085 7001_ $$00000-0002-0412-6255$$aMerchant, Thomas E$$b29
000168085 7001_ $$0P:(DE-He78)4c28e2aade5f44d8eca9dd8e97638ec8$$aKool, Marcel$$b30$$udkfz
000168085 7001_ $$00000-0003-1239-7757$$aEllison, David W$$b31
000168085 7001_ $$00000-0001-5019-0699$$aGajjar, Amar$$b32
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