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251. Neuroblastoma mRNAs Predict Outcome in Children With Stage 4 Neuroblastoma: A European HR-NBL1/SIOPEN Study

Author

Viprey, Virginie F., primary, Gregory, Walter M., additional, Corrias, Maria V., additional, Tchirkov, Andrei, additional, Swerts, Katrien, additional, Vicha, Ales, additional, Dallorso, Sandro, additional, Brock, Penelope, additional, Luksch, Roberto, additional, Valteau-Couanet, Dominique, additional, Papadakis, Vassilios, additional, Laureys, Genevieve, additional, Pearson, Andrew D., additional, Ladenstein, Ruth, additional, and Burchill, Susan A., additional

Published
2014
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252. Challenges and opportunities for international cooperative studies in pediatric hematopoeitic cell transplantation: priorities of the Westhafen Intercontinental Group.

Author

Schultz, Rudolph Kirk R, Schultz, Rudolph Kirk R, Baker, Kevin Scott, Boelens, Jaap J, Bollard, Catherine M, Egeler, R Maarten, Cowan, Mort, Ladenstein, Ruth, Lankester, Arjan, Locatelli, Franco, Lawitschka, Anita, Levine, John E, Loh, Mignon, Nemecek, Eneida, Niemeyer, Charlotte, Prasad, Vinod K, Rocha, Vanderson, Shenoy, Shalini, Strahm, Brigitte, Veys, Paul, Wall, Donna, Bader, Peter, Grupp, Stephan A, Pulsipher, Michael A, Peters, Christina, Schultz, Rudolph Kirk R, Schultz, Rudolph Kirk R, Baker, Kevin Scott, Boelens, Jaap J, Bollard, Catherine M, Egeler, R Maarten, Cowan, Mort, Ladenstein, Ruth, Lankester, Arjan, Locatelli, Franco, Lawitschka, Anita, Levine, John E, Loh, Mignon, Nemecek, Eneida, Niemeyer, Charlotte, Prasad, Vinod K, Rocha, Vanderson, Shenoy, Shalini, Strahm, Brigitte, Veys, Paul, Wall, Donna, Bader, Peter, Grupp, Stephan A, Pulsipher, Michael A, and Peters, Christina

Abstract

More than 20% of allogeneic hematopoietic cell transplantations (HCTs) are performed in children and adolescents at a large number of relatively small centers. Unlike adults, at least one-third of HCTs in children are performed for rare, nonmalignant indications. Clinical trials to improve HCT outcomes in children have been limited by small numbers and these pediatric-specific features. The need for a larger number of pediatric HCT centers to participate in trials has led to the involvement of international collaborative groups. Representatives of the Pediatric Blood and Marrow Transplant Consortium, European Group for Blood and Marrow Transplantation's Pediatric Working Group, International Berlin-Frankfurt-Munster (iBFm) Stem Cell Transplantation Committee, and Children's Oncology Group's Hematopoietic Stem Cell Transplantation Discipline Committee met on October 3, 2012, in Frankfurt, Germany to develop a consensus on the highest priorities in pediatric HCT. In addition, it explored the creation of an international consortium to develop studies focused on HCT in children and adolescents. This meeting led to the creation of an international HCT network, dubbed the Westhafen Intercontinental Group, to develop worldwide priorities and strategies to address pediatric HCT issues. This review outlines the priorities of need as identified by this consensus group.

Published
2013

255. Delayed resection can avoid irradiation in localized embryonal rhabdomyosarcoma

Author

Dantonello, Tobias M., Schuck, Andreas, Kube, Stefanie, Greulich, Michael, Kuehnle, Ingrid, Feuchtgruber, Simone, Kosztyla, Daniel, Leuschner, Ivo, Kazanowska, Bernarda, Godzinski, Jan, Ladenstein, Ruth, Niggli, Felix, Ljungman, Gustaf, Bielack, Stefan S., Klingebiel, Thomas, Koscielniak, Ewa, Dantonello, Tobias M., Schuck, Andreas, Kube, Stefanie, Greulich, Michael, Kuehnle, Ingrid, Feuchtgruber, Simone, Kosztyla, Daniel, Leuschner, Ivo, Kazanowska, Bernarda, Godzinski, Jan, Ladenstein, Ruth, Niggli, Felix, Ljungman, Gustaf, Bielack, Stefan S., Klingebiel, Thomas, and Koscielniak, Ewa

Published
2012

256. Genome-wide promoter methylation analysis in neuroblastoma identifies prognostic methylation biomarkers

Author

Decock, Anneleen, Ongenaert, Maté, Hoebeeck, Jasmien, De Preter, Katleen, Van Peer, Gert, Van Criekinge, Wim, Ladenstein, Ruth, Schulte, Johannes Hubertus, Noguera, Rosa, Stallings, Raymond R.L., Van Damme, An, Laureys, Geneviève, Vermeulen, Joëlle, Van Maerken, Tom, Speleman, Frank, Vandesompele, Jo, Decock, Anneleen, Ongenaert, Maté, Hoebeeck, Jasmien, De Preter, Katleen, Van Peer, Gert, Van Criekinge, Wim, Ladenstein, Ruth, Schulte, Johannes Hubertus, Noguera, Rosa, Stallings, Raymond R.L., Van Damme, An, Laureys, Geneviève, Vermeulen, Joëlle, Van Maerken, Tom, Speleman, Frank, and Vandesompele, Jo

Abstract

Background: Accurate outcome prediction in neuroblastoma, which is necessary to enable the optimal choice of risk-related therapy, remains a challenge. To improve neuroblastoma patient stratification, this study aimed to identify prognostic tumor DNA methylation biomarkers.Results: To identify genes silenced by promoter methylation, we first applied two independent genome-wide methylation screening methodologies to eight neuroblastoma cell lines. Specifically, we used re-expression profiling upon 5-aza-2'-deoxycytidine (DAC) treatment and massively parallel sequencing after capturing with a methyl-CpG-binding domain (MBD-seq). Putative methylation markers were selected from DAC-upregulated genes through a literature search and an upfront methylation-specific PCR on 20 primary neuroblastoma tumors, as well as through MBD- seq in combination with publicly available neuroblastoma tumor gene expression data. This yielded 43 candidate biomarkers that were subsequently tested by high-throughput methylation-specific PCR on an independent cohort of 89 primary neuroblastoma tumors that had been selected for risk classification and survival. Based on this analysis, methylation of KRT19, FAS, PRPH, CNR1, QPCT, HIST1H3C, ACSS3 and GRB10 was found to be associated with at least one of the classical risk factors, namely age, stage or MYCN status. Importantly, HIST1H3C and GNAS methylation was associated with overall and/or event-free survival.Conclusions: This study combines two genome-wide methylation discovery methodologies and is the most extensive validation study in neuroblastoma performed thus far. We identified several novel prognostic DNA methylation markers and provide a basis for the development of a DNA methylation-based prognostic classifier in neuroblastoma. © 2012 Decock et al. licensee BioMed Central Ltd., SCOPUS: ar.j, info:eu-repo/semantics/published

Published
2012

257. SURVIVAL AFTER RECURRENCE OF METASTATIC RHAFIDOMYOSARCOMA - AN UNPROBABLE, BUT NOT IMPOSSIBLE EVENT IN THE EXPERIENCE OF THE COOPERATIVE WEICHTEILSARKOM STUDIENGRUPPE (CWS)

Author

Dantonello, Tobias, Handgretinger, Rupert, Kosztyla, Daniel, Kube, Stefanie, Leuschners, Ivo, Boelling, Tobias, Bielack, Stefan S., Ladenstein, Ruth, Niggli, Felix, Kazanowska, Bernarda, Ljungman, Gustaf, Treuner, Joern, Klingebiel, Thomas, Koscielniak, Ewa, Dantonello, Tobias, Handgretinger, Rupert, Kosztyla, Daniel, Kube, Stefanie, Leuschners, Ivo, Boelling, Tobias, Bielack, Stefan S., Ladenstein, Ruth, Niggli, Felix, Kazanowska, Bernarda, Ljungman, Gustaf, Treuner, Joern, Klingebiel, Thomas, and Koscielniak, Ewa

Published
2010

258. Excellent outcome with reduced treatment for infants with disseminated neuroblastoma without MYCN gene amplification.

Author

UCL - MD/GYPE - Département de gynécologie, d'obstétrique et de pédiatrie, UCL - (SLuc) Centre du cancer, UCL - (SLuc) Service d'hématologie et d'oncologie pédiatrique, De Bernardi, Bruno, Gerrard, Mary, Boni, Luca, Rubie, Hervé, Cañete, Adela, Di Cataldo, Andrea, Castel, Victoria, Forjaz de Lacerda, Ana, Ladenstein, Ruth, Ruud, Ellen, Brichard, Bénédicte, Couturier, Jerome, Ellershaw, Caroline, Munzer, Caroline, Bruzzi, Paolo, Michon, Jean, Pearson, Andrew D J, UCL - MD/GYPE - Département de gynécologie, d'obstétrique et de pédiatrie, UCL - (SLuc) Centre du cancer, UCL - (SLuc) Service d'hématologie et d'oncologie pédiatrique, De Bernardi, Bruno, Gerrard, Mary, Boni, Luca, Rubie, Hervé, Cañete, Adela, Di Cataldo, Andrea, Castel, Victoria, Forjaz de Lacerda, Ana, Ladenstein, Ruth, Ruud, Ellen, Brichard, Bénédicte, Couturier, Jerome, Ellershaw, Caroline, Munzer, Caroline, Bruzzi, Paolo, Michon, Jean, and Pearson, Andrew D J

Abstract

PURPOSE: On the assumption that most infants with disseminated neuroblastoma without MYCN amplification (MYCNA) have a favorable prognosis, two concomitant prospective trials were started in which chemotherapy was limited to patients presenting life- or organ-threatening symptoms or overt metastases to skeleton, lung, or CNS. Surgery was to be performed only in the absence of surgical risk factors. PATIENTS AND METHODS: One hundred seventy infants with disseminated neuroblastoma without MYCNA, diagnosed between June 1999 and June 2004 in nine European countries were eligible for either of the two studies. Trial 99.2 included all stage 4S infants and those with stage 4 with a primary tumor infiltrating across the midline or positive skeletal scintigraphy who were to be observed in absence of symptoms. Trial 99.3 included infants with overt metastases to the skeleton, lung, and CNS to be treated with a minimum of four chemotherapy courses. RESULTS: The 125 infants treated on trial 99.2 had a 2-year overall survival (OS) of 97.6% with no difference between asymptomatic and symptomatic patients (97.7% v 97.3%), patients without or with unresectable primary tumors (96.8% v 100%), and patients without or with positive skeletal scintigraphy without radiologic abnormalities (97.2% v 100%). The 45 infants treated on trial 99.3 had a 2-year OS of 95.6%. No patients died of surgery- or chemotherapy-related complications. CONCLUSION: Infants with disseminated disease without MYCNA have excellent survival with minimal or no treatment. Asymptomatic infants with an unresectable primary tumor or positive skeletal scintigraphy without radiologic abnormalities may undergo observation alone.

Published
2009

260. Irinotecan and temozolomide combined with dasatinib and rapamycin for patients with relapsed or refractory neuroblastoma: Results of the prospective randomized RIST trial.

Author

Corbacioglu, Selim, Lode, Holger N., Jakob, Marcus, Suttorp, Meinolf, Escherich, Gabriele, Gruhn, Bernd, Ellinger, Susanne, Simon, Thorsten, Lang, Peter, Rohde, Marius, Zeman, Florian, Debatin, Klaus-Michael, Beilken, Andreas, Heiss, Peter, Hellwig, Dirk, Riemenschneider, Markus, Ladenstein, Ruth Lydia, Bochennek, Konrad, Eggert, Angelika, and Foell, Juergen

Published
2023
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261. High risk neuroblastoma (HR-NB) with MNA and age <18 months: Results from the HR-NBL1/SIOPEN trial.

Author

Ladenstein, Ruth Lydia, Poetschger, Ulrike, Valteau Couanet, Dominique, Ash, Shifra, Beck Popovic, Maja, Balwierz, Walentyna, Boterberg, Tom, Chan, Godfrey Chi-Fung, Elliott, Martin, Kogner, Per, Lode, Holger N., Laureys, Genevieve, Luksch, Roberto, Malis, Josef, Owens, Cormac, Papadakis, Vassilios, Sarnacki, Sabine, Taschner-Mandl, Sabine, Schleiermacher, Gudrun, and Canete, Adela

Published
2023
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263. Challenges and Opportunities for International Cooperative Studies in Pediatric Hematopoeitic Cell Transplantation: Priorities of the Westhafen Intercontinental Group

Author

Schultz, Kirk R., primary, Baker, Kevin Scott, additional, Boelens, Jaap J., additional, Bollard, Catherine M., additional, Egeler, R. Maarten, additional, Cowan, Mort, additional, Ladenstein, Ruth, additional, Lankester, Arjan, additional, Locatelli, Franco, additional, Lawitschka, Anita, additional, Levine, John E., additional, Loh, Mignon, additional, Nemecek, Eneida, additional, Niemeyer, Charlotte, additional, Prasad, Vinod K., additional, Rocha, Vanderson, additional, Shenoy, Shalini, additional, Strahm, Brigitte, additional, Veys, Paul, additional, Wall, Donna, additional, Bader, Peter, additional, Grupp, Stephan A., additional, Pulsipher, Michael A., additional, and Peters, Christina, additional

Published
2013
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265. Standardisation of operating procedures for the detection of minimal disease by QRT-PCR in children with neuroblastoma : Quality assurance on behalf of SIOPEN-R-NET

Author

Viprey, Virginie F, Corrias, Maria V, Kågedal, Bertil, Oltra, Silvestre, Swerts, Katrien, Vicha, Ales, Ladenstein, Ruth, Burchill, Susan A, Viprey, Virginie F, Corrias, Maria V, Kågedal, Bertil, Oltra, Silvestre, Swerts, Katrien, Vicha, Ales, Ladenstein, Ruth, and Burchill, Susan A

Published
2007
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267. IT Infrastructure for Merging Data from Different Clinical Trials and Across Independent Research Networks.

Author

HAYN, Dieter, FALGENHAUER, Markus, KROPF, Martin, NITZLNADER, Michael, WELTE, Stefan, EBNER, Hubert, LADENSTEIN, Ruth, SCHLEIERMACHER, Gudrun, HERO, Barbara, and SCHREIER, Günter

Abstract

Opsoclonus Myoclonus Syndrome (OMS) is a rare disease in children which is often associated with neuroblastoma and, therefore, requires treatment by pediatric neurologists and oncologists. The ongoing OMS trial investigates questions related to OMS and potentially underlying neuroblastomas. To support this trial with an adequate IT infrastructure, linkage of neuroblastoma research databases with the OMS electronic data capture (EDC) system was required. Therefore, an EDC system for the OMS trial was developed and integrated into the research infrastructure of the European Network for Cancer Research in Children and Adolescents (ENCCA) project. Application of ENNCA's pseudonymization concept enabled linkage of the OMS trial with neuroblastoma trials from two different scientific societies, while being compliant with current data protection regulations. Linkage of the neurological and the oncological domain could successfully be demonstrated and a promising concept for secondary use of the data of both domains has been developed, proofing the broad potential of the concepts for cross-domain research as promoted in the ENCCA project. [ABSTRACT FROM AUTHOR]

Published
2016
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268. Electronic Patient Reported Outcomes in Paediatric Oncology - Applying Mobile and Near Field Communication Technology.

Author

DUREGGER, Katharina, HAYN, Dieter, NITZLNADER, Michael, KROPF, Martin, FALGENHAUER, Markus, LADENSTEIN, Ruth, and SCHREIER, Günter

Abstract

Background: Electronic Patient Reported Outcomes (ePRO) gathered using telemonitoring solutions might be a valuable source of information in rare cancer research. Objectives: The objective of this paper was to develop a concept and implement a prototype for introducing ePRO into the existing neuroblastoma research network by applying Near Field Communication and mobile technology. Methods: For physicians, an application was developed for registering patients within the research network and providing patients with an ID card and a PIN for authentication when transmitting telemonitoring data to the Electronic Data Capture system OpenClinica. For patients, a previously developed telemonitoring system was extended by a Simple Object Access Protocol (SOAP) interface for transmitting nine different health parameters and toxicities. Results: The concept was fully implemented on the front-end side. The developed application for physicians was prototypically implemented and the mobile application of the telemonitoring system was successfully connected to OpenClinica. Future work will focus on the implementation of the back-end features. [ABSTRACT FROM AUTHOR]

Published
2016
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269. Interoperability Architecture for a Paediatric Oncology European Reference Network.

Author

NITZLNADER, Michael, CANETE NIETO, Adela, RIBELLES, Antonio Juan, BRUNMAIR, Barbara, LADENSTEIN, Ruth, and SCHREIER, Günter

Abstract

With the Directive 2011/24/EU on patients' rights in cross-border healthcare and the related delegated decisions, the European Commission defined a legal framework on how healthcare shall be organised by European Union (EU) member states (MS) where patients can move beyond the borders of their home country. Among other aspects, Article 12 of the directive is concerned with supporting MS with the development of so called European Reference Networks (ERN), dedicated to the treatment of "patients with a medical condition requiring a particular concentration of expertise in medical domains where expertise is rare". In the "European Expert Paediatric Oncology Reference Network for Diagnostics and Treatment" (ExPO-r-Net) project, the establishment of such an ERN in the domain of Paediatric Oncology is currently piloted. The present paper describes the high level use cases, the main requirements and a corresponding interoperability architecture capable to serve as the necessary IT platform to facilitate cross-border health data exchange. [ABSTRACT FROM AUTHOR]

Published
2016
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270. Piloting the European Unified Patient Identity Management (EUPID) Concept to Facilitate Secondary Use of Neuroblastoma Data from Clinical Trials and Biobanking.

Author

EBNER, Hubert, HAYN, Dieter, FALGENHAUER, Markus, NITZLNADER, Michael, SCHLEIERMACHER, Gudrun, HAUPT, Riccardo, ERMINIO, Giovanni, DEFFERRARI, Raffaella, MAZZOCCO, Katia, KOHLER, Jan, TONINI, Gian Paolo, LADENSTEIN, Ruth, and SCHREIER, Guenter

Abstract

Data from two contexts, i.e. the European Unresectable Neuroblastoma (EUNB) clinical trial and results from comparative genomic hybridisation (CGH) analyses from corresponding tumour samples shall be provided to existing repositories for secondary use. Utilizing the European Unified Patient IDentity Management (EUPID) as developed in the course of the ENCCA project, the following processes were applied to the data: standardization (providing interoperability), pseudonymization (generating distinct but linkable pseudonyms for both contexts), and linking both data sources. The applied procedures resulted in a joined dataset that did not contain any identifiers that would allow to backtrack the records to either data sources. This provided a high degree of privacy to the involved patients as required by data protection regulations, without preventing proper analysis. [ABSTRACT FROM AUTHOR]

Published
2016
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272. New policies to address the global burden of childhood cancers

Author

Sullivan, Richard, primary, Kowalczyk, Jerzy R, additional, Agarwal, Bharat, additional, Ladenstein, Ruth, additional, Fitzgerald, Edel, additional, Barr, Ronald, additional, Steliarova-Foucher, Eva, additional, Magrath, Ian, additional, Howard, Scott C, additional, Kruger, Mariana, additional, Valsecchi, Maria Grazia, additional, Biondi, Andrea, additional, Grundy, Paul, additional, Smith, Malcolm A, additional, Adamson, Peter, additional, Vassal, Gilles, additional, and Pritchard-Jones, Kathy, additional

Published
2013
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274. Genome-wide promoter methylation analysis in neuroblastoma identifies prognostic methylation biomarkers

Author

Decock, Anneleen, primary, Ongenaert, Maté, additional, Hoebeeck, Jasmien, additional, De Preter, Katleen, additional, Van Peer, Gert, additional, Van Criekinge, Wim, additional, Ladenstein, Ruth, additional, Schulte, Johannes H, additional, Noguera, Rosa, additional, Stallings, Raymond L, additional, Van Damme, An, additional, Laureys, Geneviève, additional, Vermeulen, Joëlle, additional, Van Maerken, Tom, additional, Speleman, Frank, additional, and Vandesompele, Jo, additional

Published
2012
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275. The First European Interdisciplinary Ewing Sarcoma Research Summit

Author

Kovar, Heinrich, primary, Alonso, Javier, additional, Aman, Pierre, additional, Aryee, Dave N. T., additional, Ban, Jozef, additional, Burchill, Sue A., additional, Burdach, Stefan, additional, De Alava, Enrique, additional, Delattre, Olivier, additional, Dirksen, Uta, additional, Fourtouna, Argyro, additional, Fulda, Simone, additional, Helman, Lee J., additional, Herrero-Martin, David, additional, Hogendoorn, Pancras C. W., additional, Kontny, Udo, additional, Lawlor, Elizabeth R., additional, Lessnick, Stephen L., additional, Llombart-Bosch, Antonio, additional, Metzler, Markus, additional, Moriggl, Richard, additional, Niedan, Stephan, additional, Potratz, Jenny, additional, Redini, Françoise, additional, Richter, Günther H. S., additional, Riedmann, Lucia T., additional, Rossig, Claudia, additional, Schäfer, Beat W., additional, Schwentner, Raphaela, additional, Scotlandi, Katia, additional, Sorensen, Poul H., additional, Staege, Martin S., additional, Tirode, Franck, additional, Toretsky, Jeffrey, additional, Ventura, Selena, additional, Eggert, Angelika, additional, and Ladenstein, Ruth, additional

Published
2012
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276. Tolerability, response and outcome of high-risk neuroblastoma patients treated with long-term infusion of anti-GD2antibody ch14.18/CHO

Author

Mueller, Ina, Ehlert, Karoline, Endres, Stefanie, Pill, Lena, Siebert, Nikolai, Kietz, Silke, Brock, Penelope, Garaventa, Alberto, Valteau-Couanet, Dominique, Janzek, Evelyne, Hosten, Norbert, Zinke, Andreas, Barthlen, Winfried, Varol, Emine, Loibner, Hans, Ladenstein, Ruth, and Lode, Holger N.

Abstract

ABSTRACTImmunotherapy with short term infusion (STI) of monoclonal anti-GD2antibody (mAb) ch14.18 (4 × 25 mg/m2/d; 8–20 h) in combination with cytokines and 13-cis retinoic acid (RA) prolonged survival in high-risk neuroblastoma (NB) patients. Here, we investigated long-term infusion (LTI) of ch14.18 produced in Chinese hamster ovary cells (ch14.18/CHO; 10 × 10 mg/m2; 24 h) in combination with subcutaneous (s.c.) interleukin-2 (IL-2) in a single center program and report clinical response, toxicity and survival. Fifty-three high-risk NB patients received up to 6 cycles of 100 mg/m2ch14.18/CHO (d8–17) as LTI combined with 6 × 106IU/m2s.c. IL-2 (d1–5; 8–12) and 160 mg/m2oral RA (d19–32). Pain toxicity was documented with validated pain scores and intravenous (i.v.) morphine usage. Response was assessed in 37/53 evaluable patients following International Neuroblastoma Risk Group criteria. Progression-free (PFS) and overall survival (OS) was analyzed by the Kaplan-Meier method and compared to a matched historical control group from the database of AIEOP, the “Italian Pediatric Ematology and Oncology Association”. LTI of ch14.18/CHO showed acceptable toxicity profile indicated by low pain scores, reduced i.v. morphine usage and low frequency of Grade ≥3 adverse events that allowed outpatient treatment. We observed a best response rate of 40.5% (15/37; 5 CR, 10 PR), 4-year (4 y) PFS of 33.1% (observation 0.1- 4.9 y, mean: 2.2 y) and a 4 y OS of 47.7% (observation 0.27 – 5.20 y, mean: 3.6 y). Survival of the entire cohort (53/53) and the relapsed patients (29/53) was significantly improved compared to historical controls. LTI of ch14.18/CHO thus shows an acceptable toxicity profile, objective clinical responses and a strong signal of clinical efficacy in NB patients.

Published
2018
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277. Clinical and Biologic Features Predictive of Survival After Relapse of Neuroblastoma: A Report From the International Neuroblastoma Risk Group Project

Author

London, Wendy B., primary, Castel, Victoria, additional, Monclair, Tom, additional, Ambros, Peter F., additional, Pearson, Andrew D.J., additional, Cohn, Susan L., additional, Berthold, Frank, additional, Nakagawara, Akira, additional, Ladenstein, Ruth L., additional, Iehara, Tomoko, additional, and Matthay, Katherine K., additional

Published
2011
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278. Changes over three decades in outcome and the prognostic influence of age-at-diagnosis in young patients with neuroblastoma: A report from the International Neuroblastoma Risk Group Project

Author

Moroz, Veronica, primary, Machin, David, additional, Faldum, Andreas, additional, Hero, Barbara, additional, Iehara, Tomoko, additional, Mosseri, Veronique, additional, Ladenstein, Ruth, additional, Bernardi, Bruno De, additional, Rubie, Hervé, additional, Berthold, Frank, additional, Matthay, Katherine K., additional, Monclair, Tom, additional, Ambros, Peter F., additional, Pearson, Andrew D.J., additional, Cohn, Susan L., additional, and London, Wendy B., additional

Published
2011
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279. A Multilocus Technique for Risk Evaluation of Patients with Neuroblastoma

Author

Ambros, Inge M., primary, Brunner, Bettina, additional, Aigner, Gerhard, additional, Bedwell, Clare, additional, Beiske, Klaus, additional, Bénard, Jean, additional, Bown, Nick, additional, Combaret, Valerie, additional, Couturier, Jerome, additional, Defferrari, Raffaella, additional, Gross, Nicole, additional, Jeison, Marta, additional, Lunec, John, additional, Marques, Barbara, additional, Martinsson, Tommy, additional, Mazzocco, Katia, additional, Noguera, Rosa, additional, Schleiermacher, Gudrun, additional, Speleman, Frank, additional, Stallings, Ray, additional, Tonini, Gian Paolo, additional, Tweddle, Deborah A., additional, Valent, Alexander, additional, Vicha, Ales, additional, Roy, Nadine Van, additional, Villamon, Eva, additional, Ziegler, Andrea, additional, Preuner, Sandra, additional, Drobics, Mario, additional, Ladenstein, Ruth, additional, Amann, Gabriele, additional, Schuit, Robert J.L., additional, Pötschger, Ulrike, additional, and Ambros, Peter F., additional

Published
2011
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280. Excellent Outcome With Reduced Treatment in Infants With Nonmetastatic and Unresectable Neuroblastoma Without MYCN Amplification: Results of the Prospective INES 99.1

Author

Rubie, Hervé, primary, De Bernardi, Bruno, additional, Gerrard, Mary, additional, Canete, Adela, additional, Ladenstein, Ruth, additional, Couturier, Jérôme, additional, Ambros, Peter, additional, Munzer, Caroline, additional, Pearson, Andrew D.J., additional, Garaventa, Alberto, additional, Brock, Penelope, additional, Castel, Victoria, additional, Valteau-Couanet, Dominique, additional, Holmes, Keith, additional, Di Cataldo, Andrea, additional, Brichard, Bénédicte, additional, Mosseri, Véronique, additional, Marquez, Catalina, additional, Plantaz, Dominique, additional, Boni, Luca, additional, and Michon, Jean, additional

Published
2011
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281. Dose Finding Study for the Use of Subcutaneous Recombinant Interleukin-2 to Augment Natural Killer Cell Numbers in an Outpatient Setting for Stage 4 Neuroblastoma After Megatherapy and Autologous Stem-Cell Reinfusion

Author

Ladenstein, Ruth, primary, Pötschger, Ulrike, additional, Siabalis, Dimitris, additional, Garaventa, Alberto, additional, Bergeron, Christophe, additional, Lewis, Ian J., additional, Stein, Jerry, additional, Kohler, Janice, additional, Shaw, Peter J., additional, Holter, Wolfgang, additional, Pistoia, Vito, additional, and Michon, Jean, additional

Published
2011
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282. Randomized Trial of Prophylactic Granulocyte Colony-Stimulating Factor During Rapid COJEC Induction in Pediatric Patients With High-Risk Neuroblastoma: The European HR-NBL1/SIOPEN Study

Author

Ladenstein, Ruth, primary, Valteau-Couanet, Dominique, additional, Brock, Penelope, additional, Yaniv, Isaac, additional, Castel, Victoria, additional, Laureys, Geneviève, additional, Malis, Josef, additional, Papadakis, Vassilios, additional, Lacerda, Ana, additional, Ruud, Ellen, additional, Kogner, Per, additional, Garami, Miklos, additional, Balwierz, Walentyna, additional, Schroeder, Henrik, additional, Beck-Popovic, Maja, additional, Schreier, Günter, additional, Machin, David, additional, Pötschger, Ulrike, additional, and Pearson, Andrew, additional

Published
2010
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283. Primary Disseminated Multifocal Ewing Sarcoma: Results of the Euro-EWING 99 Trial

Author

Ladenstein, Ruth, primary, Pötschger, Ulrike, additional, Le Deley, Marie Cécile, additional, Whelan, Jeremy, additional, Paulussen, Michael, additional, Oberlin, Odile, additional, van den Berg, Henk, additional, Dirksen, Uta, additional, Hjorth, Lars, additional, Michon, Jean, additional, Lewis, Ian, additional, Craft, Alan, additional, and Jürgens, Heribert, additional

Published
2010
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284. Predicting outcomes for children with neuroblastoma using a multigene-expression signature: a retrospective SIOPEN/COG/GPOH study

Author

Vermeulen, Joëlle, primary, De Preter, Katleen, additional, Naranjo, Arlene, additional, Vercruysse, Liesbeth, additional, Van Roy, Nadine, additional, Hellemans, Jan, additional, Swerts, Katrien, additional, Bravo, Sophie, additional, Scaruffi, Paola, additional, Tonini, Gian Paolo, additional, De Bernardi, Bruno, additional, Noguera, Rosa, additional, Piqueras, Marta, additional, Cañete, Adela, additional, Castel, Victoria, additional, Janoueix-Lerosey, Isabelle, additional, Delattre, Olivier, additional, Schleiermacher, Gudrun, additional, Michon, Jean, additional, Combaret, Valérie, additional, Fischer, Matthias, additional, Oberthuer, André, additional, Ambros, Peter F, additional, Beiske, Klaus, additional, Bénard, Jean, additional, Marques, Bárbara, additional, Rubie, Hervé, additional, Kohler, Janice, additional, Pötschger, Ulrike, additional, Ladenstein, Ruth, additional, Hogarty, Michael D, additional, McGrady, Patrick, additional, London, Wendy B, additional, Laureys, Geneviève, additional, Speleman, Frank, additional, and Vandesompele, Jo, additional

Published
2009
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285. Excellent Outcome With Reduced Treatment for Infants With Disseminated Neuroblastoma Without MYCN Gene Amplification

Author

De Bernardi, Bruno, primary, Gerrard, Mary, additional, Boni, Luca, additional, Rubie, Hervé, additional, Cañete, Adela, additional, Di Cataldo, Andrea, additional, Castel, Victoria, additional, Forjaz de Lacerda, Ana, additional, Ladenstein, Ruth, additional, Ruud, Ellen, additional, Brichard, Benédicte, additional, Couturier, Jerome, additional, Ellershaw, Caroline, additional, Munzer, Caroline, additional, Bruzzi, Paolo, additional, Michon, Jean, additional, and Pearson, Andrew D.J., additional

Published
2009
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286. Poor Survival for Infants With MYCN-Amplified Metastatic Neuroblastoma Despite Intensified Treatment: The International Society of Paediatric Oncology European Neuroblastoma Experience

Author

Canete, Adela, primary, Gerrard, Mary, additional, Rubie, Hervé, additional, Castel, Victoria, additional, Di Cataldo, Andrea, additional, Munzer, Caroline, additional, Ladenstein, Ruth, additional, Brichard, Bénédicte, additional, Bermúdez, José D., additional, Couturier, Jerôme, additional, de Bernardi, Bruno, additional, Pearson, Andrew J., additional, and Michon, Jean, additional

Published
2009
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287. Results of the EICESS-92 Study: Two Randomized Trials of Ewing's Sarcoma Treatment—Cyclophosphamide Compared With Ifosfamide in Standard-Risk Patients and Assessment of Benefit of Etoposide Added to Standard Treatment in High-Risk Patients

Author

Paulussen, Michael, primary, Craft, Alan W., additional, Lewis, Ian, additional, Hackshaw, Allan, additional, Douglas, Carolyn, additional, Dunst, Jürgen, additional, Schuck, Andreas, additional, Winkelmann, Winfried, additional, Köhler, Gabriele, additional, Poremba, Christopher, additional, Zoubek, Andreas, additional, Ladenstein, Ruth, additional, van den Berg, Henk, additional, Hunold, Andrea, additional, Cassoni, Anna, additional, Spooner, David, additional, Grimer, Robert, additional, Whelan, Jeremy, additional, McTiernan, Anne, additional, and Jürgens, Herbert, additional

Published
2008
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288. Initial Patient Characteristics Can Predict Pattern and Risk of Relapse in Localized Rhabdomyosarcoma

Author

Dantonello, Tobias M., primary, Int-Veen, Christoph, additional, Winkler, Peter, additional, Leuschner, Ivo, additional, Schuck, Andreas, additional, Schmidt, Bernhard F., additional, Lochbuehler, Helmut, additional, Kirsch, Sylvia, additional, Hallmen, Erika, additional, Veit-Friedrich, Iris, additional, Bielack, Stefan S., additional, Niggli, Felix, additional, Kazanowska, Bernarda, additional, Ladenstein, Ruth, additional, Wiebe, Thomas, additional, Klingebiel, Thomas, additional, Treuner, Joern, additional, and Koscielniak, Ewa, additional

Published
2008
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289. The ENCCA-WP7/EuroSarc/EEC/ PROVABES/EURAMOS 3rd European Bone Sarcoma Networking Meeting/Joint Workshop of EU Bone Sarcoma Translational Research Networks; Vienna, Austria, September 24-25, 2015. Workshop Report.

Author

Copland, Christopher, Kager, Leo, Ladenstein, Ruth, Brugières, Laurence, Gaspar, Nathalie, LeDeley, Marie-Cecile, Vassal, Gilles, Dutour, Aurélie, Fagioli, Franca, Ferrari, Stefano, Picci, Piero, Scotlandi, Katia, Fleuren, Emmy, Gerrand, Craig, Gonzato, Ornella, van der Graaf, Winette, Hecker-Nolting, Stefanie, Herrero-Martin, David, Tirado, Oscar M., and Kovar, Heinrich

Subjects
SARCOMA, OSTEOSARCOMA, CONFERENCES & conventions
Abstract

This report summarizes the results of the 3rd Joint ENCCA-WP7, EuroSarc, EEC, PROVABES, and EURAMOS European Bone Sarcoma Network Meeting, which was held at the Children's Cancer Research Institute in Vienna, Austria on September 24-25, 2015. The joint bone sarcoma network meetings bring together European bone sarcoma researchers to present and discuss current knowledge on bone sarcoma biology, genetics, immunology, as well as results from preclinical investigations and clinical trials, to generate novel hypotheses for collaborative biological and clinical investigations. The ultimate goal is to further improve therapy and outcome in patients with bone sarcomas. [ABSTRACT FROM AUTHOR]

Published
2016
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290. Standardized Data Sharing in a Paediatric Oncology Research Network -A Proof-of-Concept Study.

Author

HOCHEDLINGER, Nina, NITZLNADER, Michael, FALGENHAUER, Markus, WELTE, Stefan, HAYN, Dieter, KOUMAKIS, Lefteris, POTAMIAS, George, TSIKNAKIS, Manolis, SARACENO, Davide, RINALDI, Eugenia, LADENSTEIN, Ruth, and SCHREIER, Günter

Abstract

Data that has been collected in the course of clinical trials are potentially valuable for additional scientific research questions in so called secondary use scenarios. This is of particular importance in rare disease areas like paediatric oncology. If data from several research projects need to be connected, so called Core Datasets can be used to define which information needs to be extracted from every involved source system. In this work, the utility of the Clinical Data Interchange Standards Consortium (CDISC) Operational Data Model (ODM) as a format for Core Datasets was evaluated and a web tool was developed which received Source ODM XML files and - via Extensible Stylesheet Language Transformation (XSLT) - generated standardized Core Dataset ODM XML files. Using this tool, data from different source systems were extracted and pooled for joined analysis in a proof-of-concept study, facilitating both, basic syntactic and semantic interoperability. [ABSTRACT FROM AUTHOR]

Published
2015
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292. Prognostic significance of DNA di-tetraploidy in neuroblastoma

Author

Ladenstein, Ruth, primary, Ambros, Inge M., additional, P�tschger, Ulrike, additional, Amann, Gabriele, additional, Urban, Christian, additional, Fink, Franz M., additional, Schmitt, Klaus, additional, Jones, Regina, additional, Slociak, Margot, additional, Schilling, Freimuth, additional, Ritter, J�rg, additional, Berthold, Frank, additional, Gadner, H., additional, and Ambros, Peter F., additional

Published
2001
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293. DNA methylation heterogeneity defines a disease spectrum in Ewing sarcoma

Author

Sheffield, Nathan C, Pierron, Gaelle, Klughammer, Johanna, Datlinger, Paul, Schönegger, Andreas, Schuster, Michael, Hadler, Johanna, Surdez, Didier, Guillemot, Delphine, Lapouble, Eve, Freneaux, Paul, Champigneulle, Jacqueline, Bouvier, Raymonde, Walder, Diana, Ambros, Ingeborg M, Hutter, Caroline, Sorz, Eva, Amaral, Ana T, de Álava, Enrique, Schallmoser, Katharina, Strunk, Dirk, Rinner, Beate, Liegl-Atzwanger, Bernadette, Huppertz, Berthold, Leithner, Andreas, de Pinieux, Gonzague, Terrier, Philippe, Laurence, Valérie, Michon, Jean, Ladenstein, Ruth, Holter, Wolfgang, Windhager, Reinhard, Dirksen, Uta, Ambros, Peter F, Delattre, Olivier, Kovar, Heinrich, Bock, Christoph, and Tomazou, Eleni M

Abstract

Developmental tumors in children and young adults carry few genetic alterations, yet they have diverse clinical presentation. Focusing on Ewing sarcoma, we sought to establish the prevalence and characteristics of epigenetic heterogeneity in genetically homogeneous cancers. We performed genome-scale DNA methylation sequencing for a large cohort of Ewing sarcoma tumors and analyzed epigenetic heterogeneity on three levels: between cancers, between tumors, and within tumors. We observed consistent DNA hypomethylation at enhancers regulated by the disease-defining EWS-FLI1 fusion protein, thus establishing epigenomic enhancer reprogramming as a ubiquitous and characteristic feature of Ewing sarcoma. DNA methylation differences between tumors identified a continuous disease spectrum underlying Ewing sarcoma, which reflected the strength of an EWS-FLI1 regulatory signature and a continuum between mesenchymal and stem cell signatures. There was substantial epigenetic heterogeneity within tumors, particularly in patients with metastatic disease. In summary, our study provides a comprehensive assessment of epigenetic heterogeneity in Ewing sarcoma and thereby highlights the importance of considering nongenetic aspects of tumor heterogeneity in the context of cancer biology and personalized medicine.

Published
2017
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294. Bone marrows from neuroblastoma patients: An excellent source for tumor genome analyses.

Author

Abbasi, M. Reza, Rifatbegovic, Fikret, Brunner, Clemens, Ladenstein, Ruth, Ambros, Inge M., and Ambros, Peter F.

Abstract

Neuroblastoma is the most common extra-cranial solid tumor in childhood. Presence of disseminated tumor cells (DTCs) in the bone marrow (BM) at diagnosis and at relapse is a common event in stage M neuroblastomas. Although the clinical heterogeneity of disseminated neuroblastomas is frequently associated with genomic diversity, so far, only little information exists about the genomic status of DTCs. This lack of knowledge is mainly due to the varying amount of BM infiltrating tumor cells, which is usually below 30% even at diagnosis thereby hampering systematic analyses. Thus, a valuable chance to analyze metastatic and relapse clones is, so far, completely unexploited. In this study, we show that the enrichment of tumor cells in fresh or DMSO frozen BM samples with a minimum of 0.05% or 0.1% infiltration rate, respectively, by applying magnetic bead-based technique increased the DTC content to a sufficient level to allow SNP array analyses in 49 out of 69 samples. In addition, we successfully used non-enriched BM samples with ≥30% DTCs including non-stained and immunostained cytospin and BM smear slides for SNP array analyses in 44 cases. We analyzed the genomic profile of DTCs by an ultra-high density SNP array technique with highest performance detecting all segmental chromosomal aberrations, amplified regions, acquired loss of heterozygosity events and minor aberrations affecting single genes or parts thereof. [ABSTRACT FROM AUTHOR]

Published
2015
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295. Tumour volume reduction after neoadjuvant chemotherapy impacts outcome in localised embryonal rhabdomyosarcoma.

Author

Dantonello, Tobias M., Stark, Monika, Timmermann, Beate, Fuchs, Jörg, Selle, Barbara, Linderkamp, Christin, Handgretinger, Rupert, Hagen, Rudolf, Feuchtgruber, Simone, Kube, Stefanie, Kosztyla, Daniel, Kazanowska, Bernarda, Ladenstein, Ruth, Niggli, Felix, Ljungman, Gustaf, Bielack, Stefan S., Klingebiel, Thomas, and Koscielniak, Ewa

Published
2015
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299. Neuroblastoma screening in infants postponed after the sixth month of age: A trial to reduce “overdiagnosis” and to detect cases with “unfavorable” biologic features

Author

Kerbl, Reinhold, primary, Urban, Christian E., additional, Ladenstein, Ruth, additional, Ambros, Inge M., additional, Spuller, Ekkehard, additional, Mutz, Ingomar, additional, Amann, Gabriele, additional, Kovar, Heinrich, additional, Gadner, Helmut, additional, and Ambros, Peter F., additional

Published
1997
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300. A single Centre Experience with allogeneic Stem Cell Transplantation for Severe Aplastic Anaemia in Childhood

Author

Ladenstein, Ruth, primary, Peters, Christina, additional, Minkov, Milen, additional, Emminger-Schmidmeier, Waltraud, additional, Mann, Georg, additional, Höcker, Paul, additional, Hawliczek, Robert, additional, Rosenmayr, Agathe, additional, Fink, Franz-Martin, additional, Niederwieser, Dietger, additional, and Gadner, Helmut, additional

Published
1997
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