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1. Impact of trametinib on the neuropsychological profile of NF1 patients

Author

Lalancette, Eve, Cantin, Édith, Routhier, Marie-Ève, Mailloux, Chantal, Bertrand, Marie-Claude, Kiaei, Dorsa Sadat, Larouche, Valérie, Tabori, Uri, Hawkins, Cynthia, Ellezam, Benjamin, Décarie, Jean-Claude, Théoret, Yves, Métras, Marie-Élaine, McKeown, Tara, Ospina, Luis H., Vairy, Stéphanie, Ramaswamy, Vijay, Coltin, Hallie, Sultan, Serge, Legault, Geneviève, Bouffet, Éric, Lafay-Cousin, Lucie, Hukin, Juliette, Erker, Craig, Caru, Maxime, Dehaes, Mathieu, Jabado, Nada, Perreault, Sébastien, and Lippé, Sarah

Published
2024
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3. Ependymoma

Author

Kilday, John-Paul, Hukin, Juliette, Thacker, Nirav, Tabori, Uri, Ramaswamy, Vijay, Scheinemann, Katrin, editor, and Bouffet, Eric, editor

Published
2024
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5. Targeting integrated epigenetic and metabolic pathways in lethal childhood PFA ependymomas

Author

Panwalkar, Pooja, Tamrazi, Benita, Dang, Derek, Chung, Chan, Sweha, Stefan, Natarajan, Siva Kumar, Pun, Matthew, Bayliss, Jill, Ogrodzinski, Martin P, Pratt, Drew, Mullan, Brendan, Hawes, Debra, Yang, Fusheng, Lu, Chao, Sabari, Benjamin R, Achreja, Abhinav, Heon, Jin, Animasahun, Olamide, Cieslik, Marcin, Dunham, Christopher, Yip, Stephen, Hukin, Juliette, Phillips, Joanna J, Bornhorst, Miriam, Griesinger, Andrea M, Donson, Andrew M, Foreman, Nicholas K, Garton, Hugh JL, Heth, Jason, Muraszko, Karin, Nazarian, Javad, Koschmann, Carl, Jiang, Li, Filbin, Mariella G, Nagrath, Deepak, Kool, Marcel, Korshunov, Andrey, Pfister, Stefan M, Gilbertson, Richard J, Allis, C David, Chinnaiyan, Arul M, Lunt, Sophia Y, Blüml, Stefan, Judkins, Alexander R, and Venneti, Sriram

Subjects
Cancer, Biotechnology, Genetics, Rare Diseases, Orphan Drug, Neurosciences, Brain Disorders, Development of treatments and therapeutic interventions, 5.1 Pharmaceuticals, Animals, Child, Ependymoma, Epigenesis, Genetic, Epigenomics, Histones, Humans, Metabolic Networks and Pathways, Mice, Biological Sciences, Medical and Health Sciences
Abstract

Childhood posterior fossa group A ependymomas (PFAs) have limited treatment options and bear dismal prognoses compared to group B ependymomas (PFBs). PFAs overexpress the oncohistone-like protein EZHIP (enhancer of Zeste homologs inhibitory protein), causing global reduction of repressive histone H3 lysine 27 trimethylation (H3K27me3), similar to the oncohistone H3K27M. Integrated metabolic analyses in patient-derived cells and tumors, single-cell RNA sequencing of tumors, and noninvasive metabolic imaging in patients demonstrated enhanced glycolysis and tricarboxylic acid (TCA) cycle metabolism in PFAs. Furthermore, high glycolytic gene expression in PFAs was associated with a poor outcome. PFAs demonstrated high EZHIP expression associated with poor prognosis and elevated activating mark histone H3 lysine 27 acetylation (H3K27ac). Genomic H3K27ac was enriched in PFAs at key glycolytic and TCA cycle–related genes including hexokinase-2 and pyruvate dehydrogenase. Similarly, mouse neuronal stem cells (NSCs) expressing wild-type EZHIP (EZHIP-WT) versus catalytically attenuated EZHIP-M406K demonstrated H3K27ac enrichment at hexokinase-2 and pyruvate dehydrogenase, accompanied by enhanced glycolysis and TCA cycle metabolism. AMPKα-2, a key component of the metabolic regulator AMP-activated protein kinase (AMPK), also showed H3K27ac enrichment in PFAs and EZHIP-WT NSCs. The AMPK activator metformin lowered EZHIP protein concentrations, increased H3K27me3, suppressed TCA cycle metabolism, and showed therapeutic efficacy in vitro and in vivo in patient-derived PFA xenografts in mice. Our data indicate that PFAs and EZHIP-WT–expressing NSCs are characterized by enhanced glycolysis and TCA cycle metabolism. Repurposing the antidiabetic drug metformin lowered pathogenic EZHIP, increased H3K27me3, and suppressed tumor growth, suggesting that targeting integrated metabolic/epigenetic pathways is a potential therapeutic strategy for treating childhood ependymomas.

Published
2021

6. Clinical Outcomes and Patient-Matched Molecular Composition of Relapsed Medulloblastoma.

Author

Kumar, Rahul, Smith, Kyle, Deng, Maximilian, Terhune, Colt, Robinson, Giles, Orr, Brent, Liu, Anthony, Lin, Tong, Billups, Catherine, Chintagumpala, Murali, Bowers, Daniel, Hassall, Timothy, Hansford, Jordan, Khuong-Quang, Dong, Crawford, John, Bendel, Anne, Gururangan, Sridharan, Schroeder, Kristin, Bouffet, Eric, Bartels, Ute, Fisher, Michael, Cohn, Richard, Partap, Sonia, Kellie, Stewart, McCowage, Geoffrey, Paulino, Arnold, Rutkowski, Stefan, Fleischhack, Gudrun, Dhall, Girish, Klesse, Laura, Leary, Sarah, Nazarian, Javad, Kool, Marcel, Wesseling, Pieter, Ryzhova, Marina, Zheludkova, Olga, Golanov, Andrey, McLendon, Roger, Packer, Roger, Dunham, Christopher, Hukin, Juliette, Fouladi, Maryam, Faria, Claudia, Pimentel, Jose, Walter, Andrew, Jabado, Nada, Cho, Yoon-Jae, Perreault, Sebastien, Croul, Sidney, Zapotocky, Michal, Hawkins, Cynthia, Tabori, Uri, Taylor, Michael, Pfister, Stefan, Klimo, Paul, Boop, Frederick, Ellison, David, Merchant, Thomas, Onar-Thomas, Arzu, Korshunov, Andrey, Jones, David, Gajjar, Amar, Ramaswamy, Vijay, and Northcott, Paul

Subjects
Biomarkers, Tumor, Cerebellar Neoplasms, Child, Child, Preschool, Clinical Trials as Topic, DNA Methylation, Disease Progression, Epigenome, Epigenomics, Female, High-Throughput Nucleotide Sequencing, Humans, Infant, Male, Medulloblastoma, Neoplasm Recurrence, Local, Retreatment, Time Factors, Treatment Outcome
Abstract

PURPOSE: We sought to investigate clinical outcomes of relapsed medulloblastoma and to compare molecular features between patient-matched diagnostic and relapsed tumors. METHODS: Children and infants enrolled on either SJMB03 (NCT00085202) or SJYC07 (NCT00602667) trials who experienced medulloblastoma relapse were analyzed for clinical outcomes, including anatomic and temporal patterns of relapse and postrelapse survival. A largely independent, paired molecular cohort was analyzed by DNA methylation array and next-generation sequencing. RESULTS: A total of 72 of 329 (22%) SJMB03 and 52 of 79 (66%) SJYC07 patients experienced relapse with significant representation of Group 3 and wingless tumors. Although most patients exhibited some distal disease (79%), 38% of patients with sonic hedgehog tumors experienced isolated local relapse. Time to relapse and postrelapse survival varied by molecular subgroup with longer latencies for patients with Group 4 tumors. Postrelapse radiation therapy among previously nonirradiated SJYC07 patients was associated with long-term survival. Reirradiation was only temporizing for SJMB03 patients. Among 127 patients with patient-matched tumor pairs, 9 (7%) experienced subsequent nonmedulloblastoma CNS malignancies. Subgroup (96%) and subtype (80%) stabilities were largely maintained among the remainder. Rare subgroup divergence was observed from Group 4 to Group 3 tumors, which is coincident with genetic alterations involving MYC, MYCN, and FBXW7. Subgroup-specific patterns of alteration were identified for driver genes and chromosome arms. CONCLUSION: Clinical behavior of relapsed medulloblastoma must be contextualized in terms of up-front therapies and molecular classifications. Group 4 tumors exhibit slower biological progression. Utility of radiation at relapse is dependent on patient age and prior treatments. Degree and patterns of molecular conservation at relapse vary by subgroup. Relapse tissue enables verification of molecular targets and identification of occult secondary malignancies.

Published
2021

7. Conformal Radiation Therapy for Pediatric Ependymoma, Chemotherapy for Incompletely Resected Ependymoma, and Observation for Completely Resected, Supratentorial Ependymoma

Author

Merchant, Thomas E, Bendel, Anne E, Sabin, Noah D, Burger, Peter C, Shaw, Dennis W, Chang, Eric, Wu, Shengjie, Zhou, Tianni, Eisenstat, David D, Foreman, Nicholas K, Fuller, Christine E, Anderson, Edwina Templeton, Hukin, Juliette, Lau, Ching C, Pollack, Ian F, Laningham, Fred H, Lustig, Robert H, Armstrong, Floyd D, Handler, Michael H, Williams-Hughes, Chris, Kessel, Sandra, Kocak, Mehmet, Ellison, David W, and Ramaswamy, Vijay

Subjects
Cancer, Rare Diseases, Pediatric Cancer, Pediatric, Clinical Research, Brain Disorders, 6.1 Pharmaceuticals, Evaluation of treatments and therapeutic interventions, Adolescent, Adult, Antineoplastic Combined Chemotherapy Protocols, Chemoradiotherapy, Child, Child, Preschool, Cytoreduction Surgical Procedures, Ependymoma, Female, Humans, Infant, Male, Progression-Free Survival, Radiotherapy, Conformal, Supratentorial Neoplasms, Transcription Factor RelA, Treatment Outcome, Young Adult, Clinical Sciences, Oncology and Carcinogenesis, Oncology & Carcinogenesis
Abstract

PURPOSE:The Children's Oncology Group trial ACNS0121 estimated event-free survival (EFS) and overall survival for children with intracranial ependymoma treated with surgery, radiation therapy, and-selectively-with chemotherapy. Treatment was administered according to tumor location, histologic grade, and extent of resection. The impacts of histologic grade, focal copy number gain on chromosome 1q, and DNA methylation profiles were studied for those undergoing surgery and immediate postoperative conformal radiation therapy (CRT). METHODS:ACNS0121 included 356 newly diagnosed patients (ages 1 to 21 years). Patients with classic supratentorial ependymoma were observed after gross total resection (GTR). Those undergoing subtotal resection received chemotherapy, second surgery, and CRT. The remaining patients received immediate postoperative CRT after near-total resection or GTR. CRT was administered with a 1.0-cm clinical target volume margin. The cumulative total dose was 59.4 Gy, except for patients who underwent GTR and were younger than age 18 months (who received 54 Gy). Patients were enrolled between October 2003 and September 2007 and were observed for 5 years. Supratentorial tumors were evaluated for RELA fusion; infratentorial tumors, for chromosome 1q gain. Classification of posterior fossa groups A and B was made by methylation profiles. RESULTS:The 5-year EFS rates were 61.4% (95% CI, 34.5% to 89.6%), 37.2% (95% CI, 24.8% to 49.6%), and 68.5% (95% CI, 62.8% to 74.2%) for observation, subtotal resection, and near-total resection/GTR groups given immediate postoperative CRT, respectively. The 5-year EFS rates differed significantly by tumor grade (P = .0044) but not by age, location, RELA fusion status, or posterior fossa A/posterior fossa B grouping. EFS was higher for patients with infratentorial tumors without 1q gain than with 1q gain (82.8% [95% CI, 74.4% to 91.2%] v 47.4% [95% CI, 26.0% to 68.8%]; P = .0013). CONCLUSION:The EFS for patients with ependymoma younger than 3 years of age who received immediate postoperative CRT and for older patients is similar. Irradiation should remain the mainstay of care for most subtypes.

Published
2019

8. Heterogeneity within the PF-EPN-B ependymoma subgroup

Author

Cavalli, Florence MG, Hübner, Jens-Martin, Sharma, Tanvi, Luu, Betty, Sill, Martin, Zapotocky, Michal, Mack, Stephen C, Witt, Hendrik, Lin, Tong, Shih, David JH, Ho, Ben, Santi, Mariarita, Emery, Lyndsey, Hukin, Juliette, Dunham, Christopher, McLendon, Roger E, Lipp, Eric S, Gururangan, Sridharan, Grossbach, Andrew, French, Pim, Kros, Johan M, van Veelen, Marie-Lise C, Rao, Amulya A Nageswara, Giannini, Caterina, Leary, Sarah, Jung, Shin, Faria, Claudia C, Mora, Jaume, Schüller, Ulrich, Alonso, Marta M, Chan, Jennifer A, Klekner, Almos, Chambless, Lola B, Hwang, Eugene I, Massimino, Maura, Eberhart, Charles G, Karajannis, Matthias A, Lu, Benjamin, Liau, Linda M, Zollo, Massimo, Ferrucci, Veronica, Carlotti, Carlos, Tirapelli, Daniela PC, Tabori, Uri, Bouffet, Eric, Ryzhova, Marina, Ellison, David W, Merchant, Thomas E, Gilbert, Mark R, Armstrong, Terri S, Korshunov, Andrey, Pfister, Stefan M, Taylor, Michael D, Aldape, Kenneth, Pajtler, Kristian W, Kool, Marcel, and Ramaswamy, Vijay

Subjects
Genetics, Cancer, Clinical Research, Rare Diseases, Human Genome, Brain Cancer, Brain Disorders, Adolescent, Adult, Age Factors, Child, Cohort Studies, DNA Copy Number Variations, DNA Methylation, Ependymoma, Female, Gene Expression Profiling, Humans, Infratentorial Neoplasms, Kaplan-Meier Estimate, Male, Microarray Analysis, Middle Aged, Young Adult, Posterior fossa, Subgrouping, PFB, PFA, Clustering, Clinical Sciences, Neurosciences, Neurology & Neurosurgery
Abstract

Posterior fossa ependymoma comprise three distinct molecular variants, termed PF-EPN-A (PFA), PF-EPN-B (PFB), and PF-EPN-SE (subependymoma). Clinically, they are very disparate and PFB tumors are currently being considered for a trial of radiation avoidance. However, to move forward, unraveling the heterogeneity within PFB would be highly desirable. To discern the molecular heterogeneity within PFB, we performed an integrated analysis consisting of DNA methylation profiling, copy-number profiling, gene expression profiling, and clinical correlation across a cohort of 212 primary posterior fossa PFB tumors. Unsupervised spectral clustering and t-SNE analysis of genome-wide methylation data revealed five distinct subtypes of PFB tumors, termed PFB1-5, with distinct demographics, copy-number alterations, and gene expression profiles. All PFB subtypes were distinct from PFA and posterior fossa subependymomas. Of the five subtypes, PFB4 and PFB5 are more discrete, consisting of younger and older patients, respectively, with a strong female-gender enrichment in PFB5 (age: p = 0.011, gender: p = 0.04). Broad copy-number aberrations were common; however, many events such as chromosome 2 loss, 5 gain, and 17 loss were enriched in specific subtypes and 1q gain was enriched in PFB1. Late relapses were common across all five subtypes, but deaths were uncommon and present in only two subtypes (PFB1 and PFB3). Unlike the case in PFA ependymoma, 1q gain was not a robust marker of poor progression-free survival; however, chromosome 13q loss may represent a novel marker for risk stratification across the spectrum of PFB subtypes. Similar to PFA ependymoma, there exists a significant intertumoral heterogeneity within PFB, with distinct molecular subtypes identified. Even when accounting for this heterogeneity, extent of resection remains the strongest predictor of poor outcome. However, this biological heterogeneity must be accounted for in future preclinical modeling and personalized therapies.

Published
2018

9. Immunohistochemical analysis of H3K27me3 demonstrates global reduction in group-A childhood posterior fossa ependymoma and is a powerful predictor of outcome

Author

Panwalkar, Pooja, Clark, Jonathan, Ramaswamy, Vijay, Hawes, Debra, Yang, Fusheng, Dunham, Christopher, Yip, Stephen, Hukin, Juliette, Sun, Yilun, Schipper, Matthew J, Chavez, Lukas, Margol, Ashley, Pekmezci, Melike, Chung, Chan, Banda, Adam, Bayliss, Jill M, Curry, Sarah J, Santi, Mariarita, Rodriguez, Fausto J, Snuderl, Matija, Karajannis, Matthias A, Saratsis, Amanda M, Horbinski, Craig M, Carret, Anne-Sophie, Wilson, Beverly, Johnston, Donna, Lafay-Cousin, Lucie, Zelcer, Shayna, Eisenstat, David, Silva, Marianna, Scheinemann, Katrin, Jabado, Nada, McNeely, P Daniel, Kool, Marcel, Pfister, Stefan M, Taylor, Michael D, Hawkins, Cynthia, Korshunov, Andrey, Judkins, Alexander R, and Venneti, Sriram

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Brain Disorders, Genetics, Brain Cancer, Rare Diseases, Neurosciences, Clinical Research, Pediatric Cancer, Cancer, Clinical Trials and Supportive Activities, Pediatric, Child, Child, Preschool, Disease-Free Survival, Ependymoma, Female, Humans, Infant, Infratentorial Neoplasms, Jumonji Domain-Containing Histone Demethylases, Male, Prognosis, Registries, Survival Rate, Childhood ependymoma, Epigenetics, H3K27me3, Molecular subgrouping, Neurology & Neurosurgery
Abstract

Posterior fossa ependymomas (EPN_PF) in children comprise two morphologically identical, but biologically distinct tumor entities. Group-A (EPN_PFA) tumors have a poor prognosis and require intensive therapy. In contrast, group-B tumors (EPN_PFB) exhibit excellent prognosis and the current consensus opinion recommends future clinical trials to test the possibility of treatment de-escalation in these patients. Therefore, distinguishing these two tumor subtypes is critical. EPN_PFA and EPN_PFB can be distinguished based on DNA methylation signatures, but these assays are not routinely available. We have previously shown that a subset of poorly prognostic childhood EPN_PF exhibits global reduction in H3K27me3. Therefore, we set out to determine whether a simple immunohistochemical assay for H3K27me3 could be used to segregate EPN_PFA from EPN_PFB tumors. We assembled a cohort of 230 childhood ependymomas and H3K27me3 immunohistochemistry was assessed as positive or negative in a blinded manner. H3K27me3 staining results were compared with DNA methylation-based subgroup information available in 112 samples [EPN_PFA (n = 72) and EPN_PFB tumors (n = 40)]. H3K27me3 staining was globally reduced in EPN_PFA tumors and immunohistochemistry showed 99% sensitivity and 100% specificity in segregating EPN_PFA from EPN_PFB tumors. Moreover, H3K27me3 immunostaining was sufficient to delineate patients with worse prognosis in two independent, non-overlapping cohorts (n = 133 and n = 97). In conclusion, immunohistochemical evaluation of H3K27me3 global reduction is an economic, easily available and readily adaptable method for defining high-risk EPN_PFA from low-risk posterior fossa EPN_PFB tumors to inform prognosis and to enable the design of future clinical trials.

Published
2017

10. Author Correction: Locoregional delivery of CAR T cells to the cerebrospinal fluid for treatment of metastatic medulloblastoma and ependymoma

Author

Donovan, Laura K., Delaidelli, Alberto, Joseph, Sujith K., Bielamowicz, Kevin, Fousek, Kristen, Holgado, Borja L., Manno, Alex, Srikanthan, Dilakshan, Gad, Ahmed Z., Van Ommeren, Randy, Przelicki, David, Richman, Cory, Ramaswamy, Vijay, Daniels, Craig, Pallota, Jonelle G., Douglas, Tajana, Joynt, Alyssa C. M., Haapasalo, Joonas, Nor, Carolina, Vladoiu, Maria C., Kuzan-Fischer, Claudia M., Garzia, Livia, Mack, Stephen C., Varadharajan, Srinidhi, Baker, Matthew L., Hendrikse, Liam, Ly, Michelle, Kharas, Kaitlin, Balin, Polina, Wu, Xiaochong, Qin, Lei, Huang, Ning, Stucklin, Ana Guerreiro, Morrissy, A. Sorana, Cavalli, Florence M. G., Luu, Betty, Suarez, Raul, De Antonellis, Pasqualino, Michealraj, Antony, Rastan, Avesta, Hegde, Meenakshi, Komosa, Martin, Sirbu, Olga, Kumar, Sachin A., Abdullaev, Zied, Faria, Claudia C., Yip, Stephen, Hukin, Juliette, Tabori, Uri, Hawkins, Cynthia, Aldape, Ken, Daugaard, Mads, Maris, John M., Sorensen, Poul H., Ahmed, Nabil, and Taylor, Michael D.

Published
2021
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11. Integrated (epi)-Genomic Analyses Identify Subgroup-Specific Therapeutic Targets in CNS Rhabdoid Tumors

Author

Torchia, Jonathon, Golbourn, Brian, Feng, Shengrui, Ho, Ching, Sin-Chan, Patrick, Vasiljevic, Alexandre, Norman, Joseph D, Guilhamon, Paul, Garzia, Livia, Agamez, Natalia R, Lu, Mei, Chan, Tiffany S, Picard, Daniel, de Antonellis, Pasqualino, Khuong-Quang, Dong-Anh, Planello, Aline C, Zeller, Constanze, Barsyte-Lovejoy, Dalia, Lafay-Cousin, Lucie, Letourneau, Louis, Bourgey, Mathieu, Yu, Man, Gendoo, Deena MA, Dzamba, Misko, Barszczyk, Mark, Medina, Tiago, Riemenschneider, Alexandra N, Morrissy, A Sorana, Ra, Young-Shin, Ramaswamy, Vijay, Remke, Marc, Dunham, Christopher P, Yip, Stephen, Ng, Ho-keung, Lu, Jian-Qiang, Mehta, Vivek, Albrecht, Steffen, Pimentel, Jose, Chan, Jennifer A, Somers, Gino R, Faria, Claudia C, Roque, Lucia, Fouladi, Maryam, Hoffman, Lindsey M, Moore, Andrew S, Wang, Yin, Choi, Seung Ah, Hansford, Jordan R, Catchpoole, Daniel, Birks, Diane K, Foreman, Nicholas K, Strother, Doug, Klekner, Almos, Bognár, Laszló, Garami, Miklós, Hauser, Péter, Hortobágyi, Tibor, Wilson, Beverly, Hukin, Juliette, Carret, Anne-Sophie, Van Meter, Timothy E, Hwang, Eugene I, Gajjar, Amar, Chiou, Shih-Hwa, Nakamura, Hideo, Toledano, Helen, Fried, Iris, Fults, Daniel, Wataya, Takafumi, Fryer, Chris, Eisenstat, David D, Scheinemann, Katrin, Fleming, Adam J, Johnston, Donna L, Michaud, Jean, Zelcer, Shayna, Hammond, Robert, Afzal, Samina, Ramsay, David A, Sirachainan, Nongnuch, Hongeng, Suradej, Larbcharoensub, Noppadol, Grundy, Richard G, Lulla, Rishi R, Fangusaro, Jason R, Druker, Harriet, Bartels, Ute, Grant, Ronald, Malkin, David, McGlade, C Jane, Nicolaides, Theodore, Tihan, Tarik, Phillips, Joanna, Majewski, Jacek, Montpetit, Alexandre, Bourque, Guillaume, Bader, Gary D, Reddy, Alyssa T, Gillespie, G Yancey, and Warmuth-Metz, Monika

Subjects
Genetics, Human Genome, Rare Diseases, Orphan Drug, Cancer, 2.1 Biological and endogenous factors, Development of treatments and therapeutic interventions, Aetiology, 5.1 Pharmaceuticals, Cell Line, Tumor, Cell Proliferation, Cell Survival, Central Nervous System Neoplasms, Chromatin, DNA Methylation, Dasatinib, Epigenesis, Genetic, Epigenomics, Humans, Mutation, Protein Kinase Inhibitors, Pyrimidines, Receptor, Platelet-Derived Growth Factor beta, Rhabdoid Tumor, SMARCB1 Protein, Teratoma, ATRT, enhancer, epigenomics, genomics, rhabdoid tumors, subgroup-specific therapeutics, Neurosciences, Oncology and Carcinogenesis, Oncology & Carcinogenesis
Abstract

We recently reported that atypical teratoid rhabdoid tumors (ATRTs) comprise at least two transcriptional subtypes with different clinical outcomes; however, the mechanisms underlying therapeutic heterogeneity remained unclear. In this study, we analyzed 191 primary ATRTs and 10 ATRT cell lines to define the genomic and epigenomic landscape of ATRTs and identify subgroup-specific therapeutic targets. We found ATRTs segregated into three epigenetic subgroups with distinct genomic profiles, SMARCB1 genotypes, and chromatin landscape that correlated with differential cellular responses to a panel of signaling and epigenetic inhibitors. Significantly, we discovered that differential methylation of a PDGFRB-associated enhancer confers specific sensitivity of group 2 ATRT cells to dasatinib and nilotinib, and suggest that these are promising therapies for this highly lethal ATRT subtype.

Published
2016

12. Canadian Consensus for Treatment of BRAF V600E Mutated Pediatric and AYA Gliomas.

Author

Erker, Craig, Vanan, Magimairajan Issai, Larouche, Valérie, Nobre, Liana, Cacciotti, Chantel, Vairy, Stéphanie, Zelcer, Shayna, Fleming, Adam, Bouffet, Eric, Jabado, Nada, Legault, Geneviève, Renzi, Samuele, McKeown, Tara, Crooks, Bruce, Thacker, Nirav, Ramaswamy, Vijay, Coltin, Hallie, Lafay-Cousin, Lucie, Cheng, Sylvia, and Hukin, Juliette

Subjects
BRAF genes, GLIOMAS, TERMINATION of treatment, YOUNG adults, TREATMENT duration
Abstract

Background: The treatment of BRAF V600E gliomas with BRAF inhibitors (BRAFis) and MEK inhibitors (MEKis) has been increasingly integrated into clinical practice for pediatric low-grade gliomas (PLGGs) and pediatric high-grade gliomas (HGGs). However, some questions remain unanswered, such as the best time to start targeted therapy, duration of treatment, and discontinuation of therapy. Given that no clinical trial has been able to address these critical questions, we developed a Canadian Consensus statement for the treatment of BRAF V600E mutated pediatric as well as adolescent and young adult (AYA) gliomas. Methods: Canadian neuro-oncologists were invited to participate in the development of this consensus. The consensus was discussed during monthly web-based national meetings, and the algorithms were revised until a consensus was achieved. Results: A total of 26 participants were involved in the development of the algorithms. Two treatment algorithms are proposed, one for the initiation of treatment and one for the discontinuation of treatment. We suggest that most patients with BRAF V600E gliomas should be treated with BRAFis ± MEKis upfront. Discontinuation of treatment can be considered in certain circumstances, and we suggest a slow wean. Conclusions: Based on expert consensus in Canada, we developed algorithms for treatment initiation of children and AYA with BRAF V600E gliomas as well as a discontinuation algorithm. [ABSTRACT FROM AUTHOR]

Published
2024
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14. Therapeutic Impact of Cytoreductive Surgery and Irradiation of Posterior Fossa Ependymoma in the Molecular Era: A Retrospective Multicohort Analysis.

Author

Ramaswamy, Vijay, Hielscher, Thomas, Mack, Stephen C, Lassaletta, Alvaro, Lin, Tong, Pajtler, Kristian W, Jones, David TW, Luu, Betty, Cavalli, Florence MG, Aldape, Kenneth, Remke, Marc, Mynarek, Martin, Rutkowski, Stefan, Gururangan, Sridharan, McLendon, Roger E, Lipp, Eric S, Dunham, Christopher, Hukin, Juliette, Eisenstat, David D, Fulton, Dorcas, van Landeghem, Frank KH, Santi, Mariarita, van Veelen, Marie-Lise C, Van Meir, Erwin G, Osuka, Satoru, Fan, Xing, Muraszko, Karin M, Tirapelli, Daniela PC, Oba-Shinjo, Sueli M, Marie, Suely KN, Carlotti, Carlos G, Lee, Ji Yeoun, Rao, Amulya A Nageswara, Giannini, Caterina, Faria, Claudia C, Nunes, Sofia, Mora, Jaume, Hamilton, Ronald L, Hauser, Peter, Jabado, Nada, Petrecca, Kevin, Jung, Shin, Massimi, Luca, Zollo, Massimo, Cinalli, Giuseppe, Bognár, László, Klekner, Almos, Hortobágyi, Tibor, Leary, Sarah, Ermoian, Ralph P, Olson, James M, Leonard, Jeffrey R, Gardner, Corrine, Grajkowska, Wieslawa A, Chambless, Lola B, Cain, Jason, Eberhart, Charles G, Ahsan, Sama, Massimino, Maura, Giangaspero, Felice, Buttarelli, Francesca R, Packer, Roger J, Emery, Lyndsey, Yong, William H, Soto, Horacio, Liau, Linda M, Everson, Richard, Grossbach, Andrew, Shalaby, Tarek, Grotzer, Michael, Karajannis, Matthias A, Zagzag, David, Wheeler, Helen, von Hoff, Katja, Alonso, Marta M, Tuñon, Teresa, Schüller, Ulrich, Zitterbart, Karel, Sterba, Jaroslav, Chan, Jennifer A, Guzman, Miguel, Elbabaa, Samer K, Colman, Howard, Dhall, Girish, Fisher, Paul G, Fouladi, Maryam, Gajjar, Amar, Goldman, Stewart, Hwang, Eugene, Kool, Marcel, Ladha, Harshad, Vera-Bolanos, Elizabeth, Wani, Khalida, Lieberman, Frank, Mikkelsen, Tom, Omuro, Antonio M, Pollack, Ian F, Prados, Michael, Robins, H Ian, and Soffietti, Riccardo

Subjects
Humans, Ependymoma, Infratentorial Neoplasms, Combined Modality Therapy, Retrospective Studies, Cohort Studies, Adolescent, Adult, Child, Child, Preschool, Infant, Female, Male, Cytoreduction Surgical Procedures, Rare Diseases, Pediatric Cancer, Cancer, Pediatric, Patient Safety, Clinical Sciences, Oncology and Carcinogenesis, Oncology & Carcinogenesis
Abstract

PurposePosterior fossa ependymoma comprises two distinct molecular variants termed EPN_PFA and EPN_PFB that have a distinct biology and natural history. The therapeutic value of cytoreductive surgery and radiation therapy for posterior fossa ependymoma after accounting for molecular subgroup is not known.MethodsFour independent nonoverlapping retrospective cohorts of posterior fossa ependymomas (n = 820) were profiled using genome-wide methylation arrays. Risk stratification models were designed based on known clinical and newly described molecular biomarkers identified by multivariable Cox proportional hazards analyses.ResultsMolecular subgroup is a powerful independent predictor of outcome even when accounting for age or treatment regimen. Incompletely resected EPN_PFA ependymomas have a dismal prognosis, with a 5-year progression-free survival ranging from 26.1% to 56.8% across all four cohorts. Although first-line (adjuvant) radiation is clearly beneficial for completely resected EPN_PFA, a substantial proportion of patients with EPN_PFB can be cured with surgery alone, and patients with relapsed EPN_PFB can often be treated successfully with delayed external-beam irradiation.ConclusionThe most impactful biomarker for posterior fossa ependymoma is molecular subgroup affiliation, independent of other demographic or treatment variables. However, both EPN_PFA and EPN_PFB still benefit from increased extent of resection, with the survival rates being particularly poor for subtotally resected EPN_PFA, even with adjuvant radiation therapy. Patients with EPN_PFB who undergo gross total resection are at lower risk for relapse and should be considered for inclusion in a randomized clinical trial of observation alone with radiation reserved for those who experience recurrence.

Published
2016

15. Locoregional delivery of CAR T cells to the cerebrospinal fluid for treatment of metastatic medulloblastoma and ependymoma

Author

Donovan, Laura K., Delaidelli, Alberto, Joseph, Sujith K., Bielamowicz, Kevin, Fousek, Kristen, Holgado, Borja L., Manno, Alex, Srikanthan, Dilakshan, Gad, Ahmed Z., Van Ommeren, Randy, Przelicki, David, Richman, Cory, Ramaswamy, Vijay, Daniels, Craig, Pallota, Jonelle G., Douglas, Tajana, Joynt, Alyssa C.M., Haapasalo, Joonas, Nor, Carolina, Vladoiu, Maria C., Kuzan-Fischer, Claudia M., Garzia, Livia, Mack, Stephen C., Varadharajan, Srinidhi, Baker, Matthew L., Hendrikse, Liam, Ly, Michelle, Kharas, Kaitlin, Balin, Polina, Wu, Xiaochong, Qin, Lei, Huang, Ning, Stucklin, Ana Guerreiro, Morrissy, A. Sorana, Cavalli, Florence M.G., Luu, Betty, Suarez, Raul, De Antonellis, Pasqualino, Michealraj, Antony, Rastan, Avesta, Hegde, Meenakshi, Komosa, Martin, Sirbu, Olga, Kumar, Sachin A., Abdullaev, Zied, Faria, Claudia C., Yip, Stephen, Hukin, Juliette, Tabori, Uri, Hawkins, Cynthia, Aldape, Ken, Daugaard, Mads, Maris, John M., Sorenson, Poul H., Ahmed, Nabil, and Taylor, Michael D.

Subjects
T cells -- Health aspects -- Methods, Medulloblastoma -- Care and treatment, Metastasis -- Care and treatment, Cellular therapy -- Methods -- Health aspects, Immunotherapy -- Methods -- Health aspects, Antigens -- Health aspects, Cerebrospinal fluid -- Health aspects -- Methods, Biological sciences, Health
Abstract

Recurrent medulloblastoma and ependymoma are universally lethal, with no approved targeted therapies and few candidates presently under clinical evaluation. Nearly all recurrent medulloblastomas and posterior fossa group A (PFA) ependymomas are located adjacent to and bathed by the cerebrospinal fluid, presenting an opportunity for locoregional therapy, bypassing the blood-brain barrier. We identify three cell-surface targets, EPHA2, HER2 and interleukin 13 receptor [alpha]2, expressed on medulloblastomas and ependymomas, but not expressed in the normal developing brain. We validate intrathecal delivery of EPHA2, HER2 and interleukin 13 receptor [alpha]2 chimeric antigen receptor T cells as an effective treatment for primary, metastatic and recurrent group 3 medulloblastoma and PFA ependymoma xenografts in mouse models. Finally, we demonstrate that administration of these chimeric antigen receptor T cells into the cerebrospinal fluid, alone or in combination with azacytidine, is a highly effective therapy for multiple metastatic mouse models of group 3 medulloblastoma and PFA ependymoma, thereby providing a rationale for clinical trials of these approaches in humans. Intraventricularly delivered monovalent and trivalent CAR T cells exhibit greater therapeutic efficacy as compared with intravenously delivered CAR T cells in medulloblastoma xenograft mouse models and show potency in ependymoma xenograft mouse models., Author(s): Laura K. Donovan [sup.1] [sup.2] , Alberto Delaidelli [sup.3] , Sujith K. Joseph [sup.4] [sup.5] , Kevin Bielamowicz [sup.4] [sup.5] , Kristen Fousek [sup.4] [sup.5] , Borja L. Holgado [...]

Published
2020
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17. Pontine gliomas a 10-year population-based study: a report from The Canadian Paediatric Brain Tumour Consortium (CPBTC)

Author

Fonseca, Adriana, Afzal, Samina, Bowes, Lynette, Crooks, Bruce, Larouche, Valerie, Jabado, Nada, Perreault, Sebastien, Johnston, Donna L., Zelcer, Shayna, Fleming, Adam, Scheinemann, Katrin, Silva, Mariana, Vanan, Magimairajan Issai, Mpofu, Chris, Wilson, Beverly, Eisenstat, David D., Lafay-Cousin, Lucie, Hukin, Juliette, Hawkins, Cynthia, Bartels, Ute, and Bouffet, Eric

Published
2020
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18. Intracranial growing teratoma syndrome (iGTS): an international case series and review of the literature

Author

Michaiel, George, Strother, Douglas, Gottardo, Nicholas, Bartels, Ute, Coltin, Hallie, Hukin, Juliette, Wilson, Beverly, Zelcer, Shayna, Hansford, Jordan R., Hassall, Timothy, AbdelBaki, Mohamed S., Cole, Kristina A., Hoffman, Lindsey, Smiley, Natasha P., Smith, Amy, Vinitsky, Anna, Vitanza, Nicholas A., Wright, Avery, Yeo, Kee K., Chow, Lionel M. L., Vanan, Magimairajan I., Dhall, Girish, Bouffet, Eric, and Lafay-Cousin, Lucie

Published
2020
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21. A phase 2 study of trametinib for patients with pediatric glioma or plexiform neurofibroma with refractory tumor and activation of the MAPK/ERK pathway: TRAM-01

Author

Perreault, Sébastien, Larouche, Valérie, Tabori, Uri, Hawkin, Cynthia, Lippé, Sarah, Ellezam, Benjamin, Décarie, Jean-Claude, Théoret, Yves, Métras, Marie-Élaine, Sultan, Serge, Cantin, Édith, Routhier, Marie-Ève, Caru, Maxime, Legault, Geneviève, Bouffet, Éric, Lafay-Cousin, Lucie, Hukin, Juliette, Erker, Craig, and Jabado, Nada

Published
2019
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22. Low Grade Glioma

Author

Scheinemann, Katrin, Hukin, Juliette, Scheinemann, Katrin, editor, and Bouffet, Eric, editor

Published
2015
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23. Ependymoma

Author

Hukin, Juliette, Kilday, John-Paul, Tabori, Uri, Scheinemann, Katrin, editor, and Bouffet, Eric, editor

Published
2015
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24. Supplementary Tables 1 - 5, Figures 1 - 6 from Personalizing the Treatment of Pediatric Medulloblastoma: Polo-like Kinase 1 as a Molecular Target in High-Risk Children

Author

Triscott, Joanna, primary, Lee, Cathy, primary, Foster, Colleen, primary, Manoranjan, Branavan, primary, Pambid, Mary Rose, primary, Berns, Rachel, primary, Fotovati, Abbas, primary, Venugopal, Chitra, primary, O'Halloran, Katrina, primary, Narendran, Aru, primary, Hawkins, Cynthia, primary, Ramaswamy, Vijay, primary, Bouffet, Eric, primary, Taylor, Michael D., primary, Singhal, Ash, primary, Hukin, Juliette, primary, Rassekh, Rod, primary, Yip, Stephen, primary, Northcott, Paul, primary, Singh, Sheila K., primary, Dunham, Christopher, primary, and Dunn, Sandra E., primary

Published
2023
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25. Supplementary Figure Legend from Personalizing the Treatment of Pediatric Medulloblastoma: Polo-like Kinase 1 as a Molecular Target in High-Risk Children

Author

Triscott, Joanna, primary, Lee, Cathy, primary, Foster, Colleen, primary, Manoranjan, Branavan, primary, Pambid, Mary Rose, primary, Berns, Rachel, primary, Fotovati, Abbas, primary, Venugopal, Chitra, primary, O'Halloran, Katrina, primary, Narendran, Aru, primary, Hawkins, Cynthia, primary, Ramaswamy, Vijay, primary, Bouffet, Eric, primary, Taylor, Michael D., primary, Singhal, Ash, primary, Hukin, Juliette, primary, Rassekh, Rod, primary, Yip, Stephen, primary, Northcott, Paul, primary, Singh, Sheila K., primary, Dunham, Christopher, primary, and Dunn, Sandra E., primary

Published
2023
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26. Molecular subgroups of atypical teratoid rhabdoid tumours in children: an integrated genomic and clinicopathological analysis

Author

Torchia, Jonathon, Picard, Daniel, Lafay-Cousin, Lucie, Hawkins, Cynthia E, Kim, Seung-Ki, Letourneau, Louis, Ra, Young-Shin, Ho, King Ching, Chan, Tiffany Sin Yu, Sin-Chan, Patrick, Dunham, Christopher P, Yip, Stephen, Ng, Ho-keung, Lu, Jian-Qiang, Albrecht, Steffen, Pimentel, José, Chan, Jennifer A, Somers, Gino R, Zielenska, Maria, Faria, Claudia C, Roque, Lucia, Baskin, Berivan, Birks, Diane, Foreman, Nick, Strother, Douglas, Klekner, Almos, Garami, Miklos, Hauser, Peter, Hortobágyi, Tibor, Bognár, Laszló, Wilson, Beverly, Hukin, Juliette, Carret, Anne-Sophie, Van Meter, Timothy E, Nakamura, Hideo, Toledano, Helen, Fried, Iris, Fults, Daniel, Wataya, Takafumi, Fryer, Chris, Eisenstat, David D, Scheineman, Katrin, Johnston, Donna, Michaud, Jean, Zelcer, Shayna, Hammond, Robert, Ramsay, David A, Fleming, Adam J, Lulla, Rishi R, Fangusaro, Jason R, Sirachainan, Nongnuch, Larbcharoensub, Noppadol, Hongeng, Suradej, Barakzai, Muhammad Abrar, Montpetit, Alexandre, Stephens, Derek, Grundy, Richard G, Schüller, Ulrich, Nicolaides, Theodore, Tihan, Tarik, Phillips, Joanna, Taylor, Michael D, Rutka, James T, Dirks, Peter, Bader, Gary D, Warmuth-Metz, Monika, Rutkowski, Stefan, Pietsch, Torsten, Judkins, Alexander R, Jabado, Nada, Bouffet, Eric, and Huang, Annie

Published
2015
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27. Outcomes of Infants and Young Children With Relapsed Medulloblastoma After Initial Craniospinal Irradiation–Sparing Approaches: An International Cohort Study

Author

Erker, Craig, primary, Mynarek, Martin, additional, Bailey, Simon, additional, Mazewski, Claire M., additional, Baroni, Lorena, additional, Massimino, Maura, additional, Hukin, Juliette, additional, Aguilera, Dolly, additional, Cappellano, Andrea M., additional, Ramaswamy, Vijay, additional, Lassaletta, Alvaro, additional, Perreault, Sébastien, additional, Kline, Cassie N., additional, Rajagopal, Revathi, additional, Michaiel, George, additional, Zapotocky, Michal, additional, Santa-Maria Lopez, Vicente, additional, La Madrid, Andres Morales, additional, Cacciotti, Chantel, additional, Sandler, Eric S., additional, Hoffman, Lindsey M., additional, Klawinski, Darren, additional, Khan, Sara, additional, Salloum, Ralph, additional, Hoppmann, Anna L., additional, Larouche, Valérie, additional, Dorris, Kathleen, additional, Toledano, Helen, additional, Gilheeney, Stephen W., additional, Abdelbaki, Mohamed S., additional, Wilson, Beverly, additional, Tsang, Derek S., additional, Knipstein, Jeffrey, additional, Oren, Michal Yalon, additional, Shah, Shafqat, additional, Murray, Jeffrey C., additional, Ginn, Kevin F., additional, Wang, Zhihong J., additional, Fleischhack, Gudrun, additional, Obrecht, Denise, additional, Tonn, Svenja, additional, Harrod, Virginia L., additional, Matheson, Kara, additional, Crooks, Bruce, additional, Strother, Douglas R., additional, Cohen, Kenneth J., additional, Hansford, Jordan R., additional, Mueller, Sabine, additional, Margol, Ashley, additional, Gajjar, Amar, additional, Dhall, Girish, additional, Finlay, Jonathan L., additional, Northcott, Paul A., additional, Rutkowski, Stefan, additional, Clifford, Steven C., additional, Robinson, Giles, additional, Bouffet, Eric, additional, and Lafay-Cousin, Lucie, additional

Published
2022
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29. Atypical teratoid rhabdoid tumor in the first year of life: the Canadian ATRT registry experience and review of the literature

Author

Fossey, Mary, Li, Haocheng, Afzal, Samina, Carret, Anne-Sophie, Eisenstat, David D., Fleming, Adam, Hukin, Juliette, Hawkins, Cynthia, Jabado, Nada, Johnston, Donna, Brown, Tania, Larouche, Valerie, Scheinemann, Katrin, Strother, Douglas, Wilson, Beverly, Zelcer, Shayna, Huang, Annie, Bouffet, Eric, and Lafay-Cousin, Lucie

Published
2017
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32. CTNI-04. TRAM-01: A PHASE 2 STUDY OF TRAMETINIB FOR PEDIATRIC PATIENTS WITH NEUROFIBROMATOSIS TYPE 1 AND PLEXIFORM NEUROFIBROMAS

Author

Kiaei, Dorsa Sadat, primary, Larouche, Valerie, additional, Décarie, Jean-Claude, additional, Tabori, Uri, additional, Hawkins, Cynthia, additional, Lippe, Sarah, additional, Ellezam, Benjamin, additional, Ospina, Luis H, additional, Theoret, Yves, additional, Desjardins, Leandra, additional, Metras, Marie-Elaine, additional, Sultan, Serge, additional, Cantin, Edith, additional, Routhier, Marie-Eve, additional, Mailloux, Chantal, additional, Bertrand, Marie-claude, additional, Caru, Maxime, additional, McKeown, Tara, additional, Vairy, Stephanie, additional, Legault, Geneviève, additional, Bouffet, Eric, additional, Ramaswamy, Vijay, additional, Coltin, Hallie, additional, Lafay-Cousin, Lucie, additional, Hukin, Juliette, additional, Erker, Craig, additional, Jabado, Nada, additional, Dehaes, Mathieu, additional, and Perreault, Sébastien, additional

Published
2022
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35. Pediatric thalamic tumors in the MRI era: a Canadian perspective

Author

Steinbok, Paul, Gopalakrishnan, Chittur Viswanathan, Hengel, Alexander R., Vitali, Aleksander M., Poskitt, Ken, Hawkins, Cynthia, Drake, James, Lamberti-Pasculli, Maria, Ajani, Olufemi, Hader, Walter, Mehta, Vivek, McNeely, P. Daniel, McDonald, Patrick J., Ranger, Adrianna, Vassilyadi, Michael, Atkinson, Jeff, Ryall, Scott, Eisenstat, David D., and Hukin, Juliette

Published
2016
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36. MEDB-49. Relapsed SHH medulloblastomas in young children. Are there alternatives to full-dose craniospinal irradiation?

Author

Erker, Craig, primary, Craig, Brandon, additional, Bailey, Simon, additional, Massimino, Maura, additional, Larouche, Valerie, additional, L Finlay, Jonathan, additional, Kline, Cassie, additional, Michaiel, George, additional, Margol, Ashley, additional, Cohen, Kenneth, additional, Cacciotti, Chantel, additional, Harrods, Virginia, additional, Doris, Kathleen, additional, AbdelBaki, Mohammed, additional, Amayiri, Nisreen, additional, Wang, Zhihong, additional, Hansford, Jordan, additional, Hukin, Juliette, additional, Salloum, Ralph, additional, Hoffman, Lindsay, additional, Muray, Jeffrey, additional, Ginn, Kevin, additional, Zapotocky, Zapotocky, additional, Baroni, Lorena, additional, Ramaswamy, Vijay, additional, Gilheens, Stephen, additional, Aguiera, Dolli, additional, Mazewski, Claire, additional, Shah, Shafqat, additional, Strother, Douglas, additional, Muller, Sabine, additional, Gajjar, Amar, additional, Northcott, Paul, additional, Clifford, Steve, additional, Robinson, Giles, additional, Bouffet, Eric, additional, and Lafay-Cousin, Lucie, additional

Published
2022
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37. NFB-08. TRAM-01: A Phase 2 study of trametinib for pediatric patients with neurofibromatosis type 1 and plexiform neurofibromas

Author

Kiaei, Dorsa Sadat, primary, Larouche, Valérie, additional, Décarie, Jean-Claude, additional, Tabori, Uri, additional, Hawkin, Cynthia, additional, Lippé, Sarah, additional, Ellezam, Benjamin, additional, Ospina, Luis H, additional, Théoret, Yves, additional, Desjardins, Léandra, additional, Métras, Marie-Élaine, additional, Sultan, Serge, additional, Cantin, Édith, additional, Routhier, Marie-Ève, additional, Mailloux, Chantal, additional, Bertrand, Marie-Claude, additional, Caru, Maxime, additional, Vairy, Stéphanie, additional, Legault, Geneviève, additional, Bouffet, Éric, additional, Ramaswamy, Vijay, additional, Coltin, Hallie, additional, Lafay-Cousin, Lucie, additional, Hukin, Juliette, additional, Erker, Craig, additional, Jabado, Nada, additional, Dehaes, Mathieu, additional, and Perreault, Sébastien, additional

Published
2022
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38. ATRT-07. Low-grade diffusely infiltrative tumor, SMARCB1-mutant: a clinical and histopathological distinct entity showing epigenetic similarity with ATRT-MYC

Author

Thomas, Christian, primary, Federico, Aniello, additional, Bens, Susanne, additional, Hellström, Mats, additional, Casar-Borota, Olivera, additional, Kordes, Uwe, additional, Neumann, Julia E, additional, Dottermusch, Matthias, additional, Rodriguez, Fausto E, additional, Lo, Andrea C, additional, Cheng, Sylvia, additional, Hendson, Glenda, additional, Hukin, Juliette, additional, Hartmann, Christian, additional, Koch, Arend, additional, Capper, David, additional, Siebert, Reiner, additional, Paulus, Werner, additional, Nemes, Karolina, additional, Johann, Pascal D, additional, Frühwald, Michael C, additional, Kool, Marcel, additional, and Hasselblatt, Martin, additional

Published
2022
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39. Low-grade diffusely infiltrative tumour (LGDIT), SMARCB1-mutant : A clinical and histopathological distinct entity showing epigenetic similarity with ATRT-MYC

Author

Hasselblatt, Martin, Thomas, Christian, Federico, Aniello, Bens, Susanne, Hellström, Mats, Casar Borota, Olivera, Kordes, Uwe, Neumann, Julia E., Dottermusch, Matthias, Rodriguez, Fausto J., Lo, Andrea C., Cheng, Sylvia, Hendson, Glenda, Hukin, Juliette, Hartmann, Christian, Koch, Arend, Capper, David, Siebert, Reiner, Paulus, Werner, Nemes, Karolina, Johann, Pascal D., Fruehwald, Michael C., Kool, Marcel, Hasselblatt, Martin, Thomas, Christian, Federico, Aniello, Bens, Susanne, Hellström, Mats, Casar Borota, Olivera, Kordes, Uwe, Neumann, Julia E., Dottermusch, Matthias, Rodriguez, Fausto J., Lo, Andrea C., Cheng, Sylvia, Hendson, Glenda, Hukin, Juliette, Hartmann, Christian, Koch, Arend, Capper, David, Siebert, Reiner, Paulus, Werner, Nemes, Karolina, Johann, Pascal D., Fruehwald, Michael C., and Kool, Marcel

Abstract

Low-grade diffusely infiltrative tumour (LGDIT), SMARCB1-mutant, is a histopathological distinct low-grade lesion encountered in older children and young adults that shows epigenetic similarity with ATRT-MYC and has the potential for malignant progression.

Published
2022
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40. Outcomes of Infants and Young Children With Relapsed Medulloblastoma After Initial Craniospinal Irradiation–Sparing Approaches: An International Cohort Study.

Author

Erker, Craig, Mynarek, Martin, Bailey, Simon, Mazewski, Claire M., Baroni, Lorena, Massimino, Maura, Hukin, Juliette, Aguilera, Dolly, Cappellano, Andrea M., Ramaswamy, Vijay, Lassaletta, Alvaro, Perreault, Sébastien, Kline, Cassie N., Rajagopal, Revathi, Michaiel, George, Zapotocky, Michal, Santa-Maria Lopez, Vicente, La Madrid, Andres Morales, Cacciotti, Chantel, and Sandler, Eric S.

Published
2023
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41. The role of resection alone in select children with intracranial ependymoma: the Canadian Pediatric Brain Tumour Consortium experience

Author

Ailon, Tamir, Dunham, Christopher, Carret, Anne-Sophie, Tabori, Uri, Mcneely, P. Daniel, Zelcer, Shayna, Wilson, Beverley, Lafay-Cousin, Lucie, Johnston, Donna, Eisenstat, David D., Silva, Marianna, Jabado, Nada, Goddard, Karen Jane, Fryer, Chris, Hendson, Glenda, Hawkins, Cynthia, Dunn, Sandra, Yip, Stephen, Singhal, Ashutosh, and Hukin, Juliette

Published
2015
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44. Low‐grade diffusely infiltrative tumour (LGDIT), SMARCB1‐mutant: A clinical and histopathological distinct entity showing epigenetic similarity with ATRT‐MYC

Author

Hasselblatt, Martin, primary, Thomas, Christian, additional, Federico, Aniello, additional, Bens, Susanne, additional, Hellström, Mats, additional, Casar‐Borota, Olivera, additional, Kordes, Uwe, additional, Neumann, Julia E., additional, Dottermusch, Matthias, additional, Rodriguez, Fausto J., additional, Lo, Andrea C., additional, Cheng, Sylvia, additional, Hendson, Glenda, additional, Hukin, Juliette, additional, Hartmann, Christian, additional, Koch, Arend, additional, Capper, David, additional, Siebert, Reiner, additional, Paulus, Werner, additional, Nemes, Karolina, additional, Johann, Pascal D., additional, Frühwald, Michael C., additional, and Kool, Marcel, additional

Published
2022
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48. CTNI-06. TRAM-01: A PHASE 2 STUDY OF TRAMETINIB FOR PATIENTS WITH PEDIATRIC GLIOMA WITH ACTIVATION OF THE MAPK/ERK PATHWAY

Author

Perreault, Sébastien, primary, Larouche, Valérie, additional, tabori, uri, additional, Hawkins, Cynthia, additional, Lippe, sarah, additional, Ellezam, Benjamin, additional, decarie, Jean-Claude, additional, Ospina, Luis H, additional, theoret, yves, additional, Desjardins, Leandra, additional, Metras, Marie-Elaine, additional, Sultan, serge, additional, Cantin, Edith, additional, Routhier, Marie-Eve, additional, Caru, Maxime, additional, Vairy, Stephanie, additional, Legault, Geneviève, additional, Bouffet, Eric, additional, Lafay-Cousin, Lucie, additional, Hukin, Juliette, additional, Erker, Craig, additional, and Jabado, Nada, additional

Published
2021
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49. SYST-04. TRAM-01: A PHASE 2 STUDY OF TRAMETINIB FOR PATIENTS WITH PEDIATRIC GLIOMA WITH ACTIVATION OF THE MAPK/ERK PATHWAY

Author

Perreault, Sébastien, primary, Larouche, Valérie, additional, Tabori, Uri, additional, Hawkins, Cynthia, additional, Lippé, Sarah, additional, Ellezam, Benjamin, additional, Decarie, Jean-Claude, additional, Ospina, Luis H, additional, Theoret, Yves, additional, Desjardins, Leandra, additional, Metras, Marie-Élaine, additional, Sultan, Serge, additional, Cantin, Edith, additional, Routhier, Marie-Eve, additional, Caru, Maxime, additional, Vairy, Stephanie, additional, Legault, Genevieve, additional, Bouffet, Eric, additional, Lafay-Cousin, Lucie, additional, Hukin, Juliette, additional, Erker, Craig, additional, and Jabado, Nada, additional

Published
2021
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50. EZH2 Expression Is a Prognostic Factor in Childhood Intracranial Ependymoma: A Canadian Pediatric Brain Tumor Consortium Study

Author

Li, Amanda M., Dunham, Christopher, Tabori, Uri, Carret, Anne-Sophie, McNeely, Daniel P., Johnston, Donna, Lafay-Cousin, Lucie, Wilson, Beverly, Eisenstat, David D., Jabado, Nada, Zelcer, Shayna, Silva, Mariana, Scheinemann, Katrin, Fryer, Christopher, Hendson, Glenda, Fotovati, Abbas, Hawkins, Cynthia, Yip, Stephen, Dunn, Sandra E., and Hukin, Juliette

Published
2015
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