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2. PB1265 “HERMES” - HEmophilia Related MEntal illnesS: A Cross-Sectional, Multicenter Study in Switzerland

Author

Kartal-Kaess, M., primary, Maier, L., additional, Kaess, M., additional, Manuela, A., additional, Trincero, A., additional, Graf, L., additional, Hengartner, H., additional, Fontana, P., additional, von der Weid, N., additional, Scheinemann, K., additional, von Mackensen, S., additional, Lerch, S., additional, Reichl, C., additional, and Kremer-Hovinga, J., additional

Published
2023
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4. Cancer burden in adolescents and young adults in Europe

Author

Trama, A, Stark, D, Bozovic-Spasojevic, I, Gaspar, N, Peccatori, F, Toss, A, Bernasconi, A, Quarello, P, Scheinemann, K, Jezdic, S, Blondeel, A, Mountzios, G, Bielack, S, Saloustros, E, Ferrari, A, Trama, A, Stark, D, Bozovic-Spasojevic, I, Gaspar, N, Peccatori, F, Toss, A, Bernasconi, A, Quarello, P, Scheinemann, K, Jezdic, S, Blondeel, A, Mountzios, G, Bielack, S, Saloustros, E, and Ferrari, A

Abstract

Background: Cancer epidemiology is unique in adolescents and young adults (AYAs; aged 15-39 years). The European Society for Medical Oncology/European Society for Paediatric Oncology (ESMO/SIOPE) AYA Working Group aims to describe the burden of cancers in AYAs in Europe and across European Union (EU) countries. Patients and methods: We used data available on the Global Cancer Observatory. We retrieved crude and age-standardised (World Standard Population) incidence and mortality rates. We reported about AYA cancer burden in Europe and between 28 EU member states. We described incidence and mortality for all cancers and for the 13 cancers most relevant to the AYA population. Results: Incidence and mortality varied widely between countries with the highest mortality observed in Eastern EU countries. Cancers of the female breast, thyroid and male testis were the most common cancers across countries followed by melanoma of skin and cancers of the cervix. Variations in cancer incidence rates across different populations may reflect different distribution of risk factors, variations in the implementation or uptake of screening as well as overdiagnosis. AYA cancer mortality disparities may be due to variation in early-stage diagnoses, different public education and awareness of cancer symptoms, different degrees of access or availability of treatment. Conclusions: Our results highlight the future health care needs and requirements for AYA-specialised services to ensure a homogeneous treatment across different countries as well as the urgency for preventive initiatives that can mitigate the increasing burden.

Published
2023

5. VARIANT ALK-FUSION POSITIVE ANAPLASTIC LARGE CELL LYMPHOMA (ALCL): A POPULATION-BASED COHORT OF THE NHL-BFM STUDY GROUP

Author

Damm-Welk, C., primary, Luedersen, J., additional, Stadt, U., additional, Richter, J., additional, Oschlies, I., additional, Klapper, W., additional, Rosenwald, A., additional, Kalinova, M., additional, Simonitsch-Klupp, I., additional, Siebert, R., additional, Zimmermann, M., additional, Alawi, M., additional, Nakel, J., additional, Scheinemann, K., additional, Knörr, F., additional, Attarbaschi, A., additional, Kabickova, E., additional, and Woessmann, W., additional

Published
2022
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7. Essential medicines for childhood cancer in Europe: a pan-European, systematic analysis by SIOPE

Author

Maria, O, Eva, B, Pamela R, K, Olga, K, Marko, O, Reineke A, S, Gilles, V, Achini, F, Balduzzi, A, Beck Popovic, M, Beishuizen, A, Bergamaschi, L, Biondi, A, Bourdeaut, F, Braicu, E, Brok, J, Brugières, L, Burke, A, Calaminus, G, Casanova, M, Choucair, M, Cleirec, M, Corbaciouglu, S, Genoveva Correa Llano, M, De Rojas, T, Domínguez Pinilla, N, Elmaraghi, C, Ferrari, A, Fossa, A, Gaspar, N, Herold, N, Karapiperi, K, Karu, M, Kjærsgaar, M, Knörr, F, Koenig, C, Kranjcec, I, Krawczyk, M, Lehmberg, K, Lehrnbecher, T, Lunesink, M, Massano, D, Matijasic, N, Merks, H, Metzler, M, Michalski, A, Minkov, M, Morland, B, Niktoreh, N, Oltenau, E, Orbach, D, Owens, C, Papachristidou, S, Pasqualini, C, Pavlovic, M, Perez Albert, P, Poyer, F, Radulovic, I, Reinhardt, D, Rebelo, J, Roser, E, Russo, I, Scheinemann, K, Schindera, C, Schrappe, M, Sehested, A, Sehouli, J, Spreafico, F, J Strauss, S, Stutterheim, J, Svojgr, K, Tzotzola, V, Van Ewijk, R, Verschuur, A, Vora, A, Woessmann, W, Zajac-Spychala, O, Zwaan, M, Otth, Maria, Brack, Eva, Kearns, Pamela R, Kozhaeva, Olga, Ocokoljic, Marko, Schoot, Reineke A, Vassal, Gilles, Federica Achini, Adriana Balduzzi, Maja Beck Popovic, Auke Beishuizen, Luca Bergamaschi, Andrea Biondi, Franck Bourdeaut, Elena Braicu, Jesper Brok, Laurence Brugières, Amos Burke, Gabriele Calaminus, Michela Casanova, Marie-Louise Choucair, Morgane Cleirec, Selim Corbaciouglu, Maria Genoveva Correa Llano, Teresa De Rojas, Nerea Domínguez Pinilla, Caroline Elmaraghi, Andrea Ferrari, Alexander Fossa, Nathalie Gaspar, Nikolas Herold, Kyriaki Karapiperi, Maarja Karu, Mimi Kjærsgaar, Fabian Knörr, Christa Koenig, Izabela Kranjcec, Malgorzata Krawczyk, Kai Lehmberg, Thomas Lehrnbecher, Maaike Lunesink, Davide Massano, Nuša Matijasic, Hans Merks, Markus Metzler, Anthony Michalski, Milen Minkov, Bruce Morland, Naghmeh Niktoreh, Elena Oltenau, Daniel Orbach, Cormac Owens, Smaragda Papachristidou, Claudia Pasqualini, Maja Pavlovic, Paula Perez Albert, Fiona Poyer, Ivana Radulovic, Dirk Reinhardt, Joana Rebelo, Eva Roser, Ida Russo, Katrin Scheinemann, Christina Schindera, Martin Schrappe, Astrid Sehested, Jalid Sehouli, Filippo Spreafico, Sandra J Strauss, Janine Stutterheim, Karel Svojgr, Vasiliki Tzotzola, Roelof Van Ewijk, Arnauld Verschuur, Ajay Vora, Willi Woessmann, Olga Zajac-Spychala, Michel Zwaan, Maria, O, Eva, B, Pamela R, K, Olga, K, Marko, O, Reineke A, S, Gilles, V, Achini, F, Balduzzi, A, Beck Popovic, M, Beishuizen, A, Bergamaschi, L, Biondi, A, Bourdeaut, F, Braicu, E, Brok, J, Brugières, L, Burke, A, Calaminus, G, Casanova, M, Choucair, M, Cleirec, M, Corbaciouglu, S, Genoveva Correa Llano, M, De Rojas, T, Domínguez Pinilla, N, Elmaraghi, C, Ferrari, A, Fossa, A, Gaspar, N, Herold, N, Karapiperi, K, Karu, M, Kjærsgaar, M, Knörr, F, Koenig, C, Kranjcec, I, Krawczyk, M, Lehmberg, K, Lehrnbecher, T, Lunesink, M, Massano, D, Matijasic, N, Merks, H, Metzler, M, Michalski, A, Minkov, M, Morland, B, Niktoreh, N, Oltenau, E, Orbach, D, Owens, C, Papachristidou, S, Pasqualini, C, Pavlovic, M, Perez Albert, P, Poyer, F, Radulovic, I, Reinhardt, D, Rebelo, J, Roser, E, Russo, I, Scheinemann, K, Schindera, C, Schrappe, M, Sehested, A, Sehouli, J, Spreafico, F, J Strauss, S, Stutterheim, J, Svojgr, K, Tzotzola, V, Van Ewijk, R, Verschuur, A, Vora, A, Woessmann, W, Zajac-Spychala, O, Zwaan, M, Otth, Maria, Brack, Eva, Kearns, Pamela R, Kozhaeva, Olga, Ocokoljic, Marko, Schoot, Reineke A, Vassal, Gilles, Federica Achini, Adriana Balduzzi, Maja Beck Popovic, Auke Beishuizen, Luca Bergamaschi, Andrea Biondi, Franck Bourdeaut, Elena Braicu, Jesper Brok, Laurence Brugières, Amos Burke, Gabriele Calaminus, Michela Casanova, Marie-Louise Choucair, Morgane Cleirec, Selim Corbaciouglu, Maria Genoveva Correa Llano, Teresa De Rojas, Nerea Domínguez Pinilla, Caroline Elmaraghi, Andrea Ferrari, Alexander Fossa, Nathalie Gaspar, Nikolas Herold, Kyriaki Karapiperi, Maarja Karu, Mimi Kjærsgaar, Fabian Knörr, Christa Koenig, Izabela Kranjcec, Malgorzata Krawczyk, Kai Lehmberg, Thomas Lehrnbecher, Maaike Lunesink, Davide Massano, Nuša Matijasic, Hans Merks, Markus Metzler, Anthony Michalski, Milen Minkov, Bruce Morland, Naghmeh Niktoreh, Elena Oltenau, Daniel Orbach, Cormac Owens, Smaragda Papachristidou, Claudia Pasqualini, Maja Pavlovic, Paula Perez Albert, Fiona Poyer, Ivana Radulovic, Dirk Reinhardt, Joana Rebelo, Eva Roser, Ida Russo, Katrin Scheinemann, Christina Schindera, Martin Schrappe, Astrid Sehested, Jalid Sehouli, Filippo Spreafico, Sandra J Strauss, Janine Stutterheim, Karel Svojgr, Vasiliki Tzotzola, Roelof Van Ewijk, Arnauld Verschuur, Ajay Vora, Willi Woessmann, Olga Zajac-Spychala, and Michel Zwaan

Abstract

Background: Shortages and unequal access to anticancer medicines for children and adolescents are a reality in Europe. The aim of the European Society for Paediatric Oncology (SIOPE) Essential Anticancer Medicines Project was to provide a list of anticancer medicines that are considered essential in the treatment of paediatric cancers to help ensure their continuous access to all children and adolescents with cancer across Europe. Methods: This pan-European project, done between Jan 20, 2020, and Feb 18, 2022, was designed to be a systematic collection and review of treatment protocols and strategies that are used to treat childhood cancer in Europe. We formed 16 working groups on the basis of paediatric cancer types, and which were based on the existing SIOPE Clinical Trial Groups. Workings groups consisted of representatives from the SIOPE Clinical Trial Groups, Young SIOPE members, and senior paediatric oncology experts. Each group collected existing treatment protocols that are used to treat the respective cancer types in Europe. Medicines from the standard group of each protocol were extracted. For medicines not on the WHO Essential Medicines List for children (EMLc) 2017, working groups did a literature search to determine whether the medicines should be defined as essential, promising, or neither essential nor promising. Each group provided an individual summary, and all medicines that were considered essential by at least one group were combined in a joint list. Findings: The working groups identified 73 treatment protocols used in Europe and defined 66 medicines as essential. For several newer medicines, such as kinase inhibitors or tisagenlecleucel, the supporting evidence was insufficient to consider them essential, so these medicines were defined as promising. 25 medicines were considered promising by at least one working group. 22 (33%) of the 66 essential and none of the promising medicines were included in the WHO EMLc 2017. The WHO EMLc 2021 included

Published
2022

9. Recommendations for the surveillance of cancer-related fatigue in childhood, adolescent, and young adult cancer survivors: a report from the International Late Effects of Childhood Cancer Guideline Harmonization Group

Author

Christen, S., Roser, K., Mulder, R.L., Ilic, A., Lie, H.C., Loonen, J.J., Mellblom, A.V., Kremer, L.C., Hudson, M.M., Constine, L.S., Skinner, R., Scheinemann, K., Marchak, J. Gilleland, and Michel, G.

Subjects
Healthcare improvement science Radboud Institute for Health Sciences [Radboudumc 18]
Abstract

Contains fulltext : 229304.pdf (Publisher’s version ) (Open Access) PURPOSE: Cancer-related fatigue (CRF) negatively affects the lives of childhood, adolescent, and young adult (CAYA) cancer survivors. We aimed to provide an evidence-based clinical practice guideline (CPG) with internationally harmonized CRF surveillance recommendations for CAYA cancer survivors diagnosed

Published
2020

10. Recommendations for ototoxicity surveillance for childhood, adolescent, and young adult cancer survivors: a report from the International Late Effects of Childhood Cancer Guideline Harmonization Group in collaboration with the PanCare Consortium

Author

Clemens, Eva, Van den Heuvel - Eibrink, Marry, Mulder, RL, Kremer, LCM (Leontien), Hudson, MM, Skinner, R, Constine, LS, Bass, JK, Kuehni, CE, Langer, T, van Dalen, EC, Bardi, E, Bonne, NX, Brock, PR, Brooks, B, Carleton, B, Caron, E, Chang, KW, Johnston, K, Knight, K, Nathan, PC, Orgel, E, Prasad, PK, Rottenberg, J, Scheinemann, K, de Vries, A.C.H., Walwyn, T, Weiss, A, Zehnhoff-Dinnesen, AA, Cohn, RJ, Landier, W, Kadan-Lottick, N, Levitt, G, Hoetink, A, Mussman, J, Princess Máxima Center for Pediatric Oncology [Utrecht, Netherlands], Erasmus MC-Sophia Hospital [Rotterdam, Netherlands], Emma Children's Hospital [Amsterdam, Netherlands] (Academic Medical Center), University of Amsterdam [Amsterdam] (UvA), St Jude Children's Research Hospital, Great North Children’s Hospital [Newcastle Upon Tyne, UK], Northern Institute for Cancer Research [Newcastle] (NICR), Newcastle University [Newcastle], University of Rochester Medical Center (URMC), Institute of Social and Preventive Medicine [Bern] (ISPM), Universität Bern [Bern] (UNIBE), University Hospital for Children and Adolescents [Lübeck, Germany], Protéomique, Réponse Inflammatoire, Spectrométrie de Masse (PRISM) - U 1192 (PRISM), Institut National de la Santé et de la Recherche Médicale (INSERM)-Université de Lille-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), Service d'Otologie et d'Otoneurologie [CHU Lille], Centre Hospitalier Universitaire de Lille (CHU de Lille), Great Ormond Street Hospital for Children NHS Foundation Trust [London, UK] (GOSHC), University of British Columbia (UBC), Stanford University, Sydney Children's hospital, Oregon Health and Science University [Portland] (OHSU), The Hospital for sick children [Toronto] (SickKids), Children’s Hospital Los Angeles [Los Angeles], University of Southern California (USC), Louisiana State University (LSU), Children's Hospital at New Orleans, Masaryk University [Brno] (MUNI), Kantonsspital Aarau [Aarau, Switzerland], University Hospital Basel [Basel], McMaster University [Hamilton, Ontario], Perth Children's Hospital [Nedlands, WA, Australia], The University of Western Australia (UWA), University of Regensburg, University Hospital Münster - Universitaetsklinikum Muenster [Germany] (UKM), University of New South Wales [Sydney] (UNSW), University of Alabama at Birmingham [ Birmingham] (UAB), SALZET, Michel, Pediatrics, Universität Bern [Bern], Université de Lille-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre Hospitalier Régional Universitaire [Lille] (CHRU Lille), and Great Ormond Street Hospital for Children NHS Foundation Trust [London, UK]

Subjects
Shunt placement, Pediatrics, medicine.medical_specialty, Adolescent, [SDV]Life Sciences [q-bio], Childhood cancer, Harmonization, Antineoplastic Agents, Platinum Compounds, Article, 03 medical and health sciences, Young Adult, 0302 clinical medicine, Ototoxicity, SDG 3 - Good Health and Well-being, Cancer Survivors, Neoplasms, otorhinolaryngologic diseases, Medicine, Humans, Young adult, 030223 otorhinolaryngology, Child, Evidence-Based Medicine, business.industry, Cancer, Guideline, medicine.disease, 3. Good health, [SDV] Life Sciences [q-bio], Oncology, 030220 oncology & carcinogenesis, Population Surveillance, Cranial Irradiation, business, Delivery of Health Care
Abstract

International audience; Childhood, adolescent, and young adult (CAYA) cancer survivors treated with platinum-based drugs, head or brain radiotherapy, or both have an increased risk of ototoxicity (hearing loss, tinnitus, or both). To ensure optimal care and reduce consequent problems—such as speech and language, social–emotional development, and learning difficulties—for these CAYA cancer survivors, clinical practice guidelines for monitoring ototoxicity are essential. The implementation of surveillance across clinical settings is hindered by differences in definitions of hearing loss, recommendations for surveillance modalities, and remediation. To address these deficiencies, the International Guideline Harmonization Group organised an international multidisciplinary panel, including 32 experts from ten countries, to evaluate the quality of evidence for ototoxicity following platinum-based chemotherapy and head or brain radiotherapy, and formulate and harmonise ototoxicity surveillance recommendations for CAYA cancer survivors.

Published
2019
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11. Long-term follow-up after childhood cancer in Switzerland: a position statement from the pediatric Swiss LTFU working group

Author

Tinner, E.M., Gumy Pause, Fabienne, Diezi, M., Bergsträsser, E., Hengartner, H., Eisenreich, B., Brazzola, P., von der Weid, N., Tomášiková, Z., and Scheinemann, K.

Subjects
ddc:618
Abstract

With better risk assessments, improvements in treatments and optimized supportive care more than 87% of the children with cancer become long-term survivors in Switzerland. Many studies have demonstrated that this enlarging population has a lifelong increased risk of morbidity and mortality associated with the underlying disease and the therapies received. Altogether, two thirds of the survivors will present at least one chronic health condition during their life and for one-third of them, the condition will be severe or life threatening as late effect of their treatment. In this context, a structured long-term follow-up is needed. The aim of this article is to give a global vision of the current situation in Switzerland concerning the different care models proposed to childhood cancer survivors and to provide recommendations for appropriate transition and long-term follow-up care.

Published
2019

13. Validation of questionnaire-reported chest wall abnormalities with a telephone interview in Swiss childhood cancer survivors.

Author

Kasteler, Rahel, Lichtensteiger, Christa, Schindera, Christina, Ansari, Marc, Kuehni, Claudia E., for the Swiss Pediatric Oncology Group (SPOG) Scientific Committee, Rössler, J., Ansari, M., Beck Popovic, M., Brazzola, P., Greiner, J., Niggli, F., Hengartner, H., Kuehni, C., Schilling, F., Scheinemann, K., von der Weid, N., Gerber, N., and Swiss Pediatric Oncology Group (SPOG) Scientific Committee

Subjects
CENTRAL nervous system tumors, CHILDHOOD cancer, CANCER survivors, TELEPHONE interviewing, PHYSICIANS, HUMAN abnormalities, CHEST (Anatomy), RESEARCH evaluation, INTERVIEWING, RESEARCH funding
Abstract

Background: Chest wall abnormalities are a poorly studied complication after treatment for childhood cancer. Chest wall abnormalities are not well-described in the literature, and little is known on the impact on daily life of survivors.Methods: We investigated prevalence and risk factors of chest wall abnormalities in childhood cancer survivors in a nationwide, population-based cohort study (Swiss Childhood Cancer Survivor Study) with a questionnaire survey. We then interviewed a nested sample of survivors to validate types of chest wall abnormalities and understand their impact on the daily life of survivors.Results: Forty-eight of 2382 (95%CI 2-3%) survivors reported a chest wall abnormality. Risk factors were older age at cancer diagnosis (16-20 years; OR 2.5, 95%CI 1.0-6.1), lymphoma (OR 3.8, 95%CI 1.2-11.4), and central nervous system tumors (OR 9.5, 95%CI 3.0-30.1) as underlying disease, and treatment with thoracic radiotherapy (OR 2.0, 95%CI 1.0-4.2), surgery to the chest (OR 4.5, 95%CI 1.8-11.5), or chemotherapy (OR 2.9, 95%CI 1.0-8.1). The nature of the chest wall abnormalities varied and included thoracic wall deformities (30%), deformations of the spine (5%) or both (55%), and scars (10%). Chest wall abnormalities affected daily life in two thirds (13/20) of those who reported these problems and necessitated medical attention for 15 (75%) survivors.Conclusion: It is important that, during follow-up care, physicians pay attention to chest wall abnormalities, which are rare late effects of cancer treatment, but can considerably affect the well-being of cancer survivors. [ABSTRACT FROM AUTHOR]

Published
2021
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14. Household income and risk-of-poverty of parents of long-term childhood cancer survivors

Author

Mader, L., Roser, K., Baenziger, J., Tinner, E.M., Scheinemann, K., Kuehni, C.E., Michel, G., and for the Swiss Paediatric Oncology Group (SPOG), .

Subjects
610 Medicine & health, 360 Social problems & social services
Abstract

Background: Taking care of children diagnosed with cancer affects parents’ professional life and may place the family at risk-of-poverty. We aimed to (i) compare the household income and risk-of-poverty of parents of childhood cancer survivors (CCS) to parents of the general population, and (ii) identify sociodemographic and cancer-related factors associated with risk-of-poverty. \ud \ud Methods: As part of the Swiss Childhood Cancer Survivor Study, we sent a questionnaire to parents of CCS aged 5–15 years, who survived ≥5 years after diagnosis. Information on parents of the general population came from the Swiss Household Panel (parents with ≥1 child aged 5–15 years). Risk-of-poverty was defined as having a monthly household income of

Published
2017

15. Bayesian spatial modelling of childhood cancer incidence in Switzerland using exact point data: a nationwide study during 1985–2015.

Author

Konstantinoudis, Garyfallos, Schuhmacher, Dominic, Ammann, Roland A., Diesch, Tamara, Kuehni, Claudia E., Spycher, Ben D., for the Swiss Paediatric Oncology Group, Ammann, R. A., Scheinemann, K., Ansari, M., Popovic, M. Beck, Brazzola, P., Greiner, J., Grotzer, M., Hengartner, H., Kuehne, T., Rössler, J., Niggli, F., Schilling, F., and von der Weid, N.

Subjects
CHILDHOOD cancer, CENTRAL nervous system cancer, BACKGROUND radiation, JUVENILE diseases, CENTRAL nervous system, ENVIRONMENTAL exposure prevention
Abstract

Background: The aetiology of most childhood cancers is largely unknown. Spatially varying environmental factors such as traffic-related air pollution, background radiation and agricultural pesticides might contribute to the development of childhood cancer. This study is the first investigation of the spatial disease mapping of childhood cancers using exact geocodes of place of residence. Methods: We included 5947 children diagnosed with cancer in Switzerland during 1985–2015 at 0–15 years of age from the Swiss Childhood Cancer Registry. We modelled cancer risk using log-Gaussian Cox processes and indirect standardisation to adjust for age and year of diagnosis. We examined whether the spatial variation of risk can be explained by modelled ambient air concentration of NO2, modelled exposure to background ionising radiation, area-based socio-economic position (SEP), linguistic region, duration in years of general cancer registration in the canton or degree of urbanisation. Results: For all childhood cancers combined, the posterior median relative risk (RR), compared to the national level, varied by location from 0.83 to 1.13 (min to max). Corresponding ranges were 0.96 to 1.09 for leukaemia, 0.90 to 1.13 for lymphoma, and 0.82 to 1.23 for central nervous system (CNS) tumours. The covariates considered explained 72% of the observed spatial variation for all cancers, 81% for leukaemia, 82% for lymphoma and 64% for CNS tumours. There was weak evidence of an association of CNS tumour incidence with modelled exposure to background ionising radiation (RR per SD difference 1.17; 0.98–1.40) and with SEP (1.6; 1.00–1.13). Conclusion: Of the investigated diagnostic groups, childhood CNS tumours showed the largest spatial variation. The selected covariates only partially explained the observed variation of CNS tumours suggesting that other environmental factors also play a role. [ABSTRACT FROM AUTHOR]

Published
2020
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18. A.03 Analyses of surgical and MRI factors associated with cerebellar mutism

Author

Sergeant, A, primary, Kameda-Smith, MM, additional, Manoranjan, B, additional, Kumar, B, additional, Duckworth, J, additional, Petrelli, T, additional, Savage, K, additional, Ajani, O, additional, Yarascavitch, B, additional, Samaan, C, additional, Scheinemann, K, additional, Alyman, C, additional, Almenawer, S, additional, Farrokhyar, F, additional, Fleming, AJ, additional, Singh, SK, additional, and Stein, N, additional

Published
2017
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19. Integrated (epi)-Genomic Analyses Identify Subgroup-Specific Therapeutic Targets in CNS Rhabdoid Tumors.

Author

Torchia, J, Golbourn, B, Feng, S, Ho, KC, Sin-Chan, P, Vasiljevic, A, Norman, JD, Guilhamon, P, Garzia, L, Agamez, NR, Lu, M, Chan, TS, Picard, D, de Antonellis, P, Khuong-Quang, D-A, Planello, AC, Zeller, C, Barsyte-Lovejoy, D, Lafay-Cousin, L, Letourneau, L, Bourgey, M, Yu, M, Gendoo, DMA, Dzamba, M, Barszczyk, M, Medina, T, Riemenschneider, AN, Morrissy, AS, Ra, Y-S, Ramaswamy, V, Remke, M, Dunham, CP, Yip, S, Ng, H-K, Lu, J-Q, Mehta, V, Albrecht, S, Pimentel, J, Chan, JA, Somers, GR, Faria, CC, Roque, L, Fouladi, M, Hoffman, LM, Moore, AS, Wang, Y, Choi, SA, Hansford, JR, Catchpoole, D, Birks, DK, Foreman, NK, Strother, D, Klekner, A, Bognár, L, Garami, M, Hauser, P, Hortobágyi, T, Wilson, B, Hukin, J, Carret, A-S, Van Meter, TE, Hwang, EI, Gajjar, A, Chiou, S-H, Nakamura, H, Toledano, H, Fried, I, Fults, D, Wataya, T, Fryer, C, Eisenstat, DD, Scheinemann, K, Fleming, AJ, Johnston, DL, Michaud, J, Zelcer, S, Hammond, R, Afzal, S, Ramsay, DA, Sirachainan, N, Hongeng, S, Larbcharoensub, N, Grundy, RG, Lulla, RR, Fangusaro, JR, Druker, H, Bartels, U, Grant, R, Malkin, D, McGlade, CJ, Nicolaides, T, Tihan, T, Phillips, J, Majewski, J, Montpetit, A, Bourque, G, Bader, GD, Reddy, AT, Gillespie, GY, Warmuth-Metz, M, Rutkowski, S, Tabori, U, Lupien, M, Brudno, M, Schüller, U, Pietsch, T, Judkins, AR, Hawkins, CE, Bouffet, E, Kim, S-K, Dirks, PB, Taylor, MD, Erdreich-Epstein, A, Arrowsmith, CH, De Carvalho, DD, Rutka, JT, Jabado, N, Huang, A, Torchia, J, Golbourn, B, Feng, S, Ho, KC, Sin-Chan, P, Vasiljevic, A, Norman, JD, Guilhamon, P, Garzia, L, Agamez, NR, Lu, M, Chan, TS, Picard, D, de Antonellis, P, Khuong-Quang, D-A, Planello, AC, Zeller, C, Barsyte-Lovejoy, D, Lafay-Cousin, L, Letourneau, L, Bourgey, M, Yu, M, Gendoo, DMA, Dzamba, M, Barszczyk, M, Medina, T, Riemenschneider, AN, Morrissy, AS, Ra, Y-S, Ramaswamy, V, Remke, M, Dunham, CP, Yip, S, Ng, H-K, Lu, J-Q, Mehta, V, Albrecht, S, Pimentel, J, Chan, JA, Somers, GR, Faria, CC, Roque, L, Fouladi, M, Hoffman, LM, Moore, AS, Wang, Y, Choi, SA, Hansford, JR, Catchpoole, D, Birks, DK, Foreman, NK, Strother, D, Klekner, A, Bognár, L, Garami, M, Hauser, P, Hortobágyi, T, Wilson, B, Hukin, J, Carret, A-S, Van Meter, TE, Hwang, EI, Gajjar, A, Chiou, S-H, Nakamura, H, Toledano, H, Fried, I, Fults, D, Wataya, T, Fryer, C, Eisenstat, DD, Scheinemann, K, Fleming, AJ, Johnston, DL, Michaud, J, Zelcer, S, Hammond, R, Afzal, S, Ramsay, DA, Sirachainan, N, Hongeng, S, Larbcharoensub, N, Grundy, RG, Lulla, RR, Fangusaro, JR, Druker, H, Bartels, U, Grant, R, Malkin, D, McGlade, CJ, Nicolaides, T, Tihan, T, Phillips, J, Majewski, J, Montpetit, A, Bourque, G, Bader, GD, Reddy, AT, Gillespie, GY, Warmuth-Metz, M, Rutkowski, S, Tabori, U, Lupien, M, Brudno, M, Schüller, U, Pietsch, T, Judkins, AR, Hawkins, CE, Bouffet, E, Kim, S-K, Dirks, PB, Taylor, MD, Erdreich-Epstein, A, Arrowsmith, CH, De Carvalho, DD, Rutka, JT, Jabado, N, and Huang, A

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

20. ALK‐POSITIVE ANAPLASTIC LARGE CELL LYMPHOMA WITH VARIANT ALK‐FUSION PARTNER: A POPULATION‐BASED ANALYSES OF THE NHL‐BFM STUDY GROUP.

Author

Damm‐Welk, C., Luedersen, J., Stadt, U. zur, Richter, J., Oschlies, I., Klapper, W., Rosenwald, A., Kalinova, M., Simonitsch‐Klupp, I., Siebert, R., Zimmermann, M., Alawi, M., Scheinemann, K., Knörr, F., Attarbaschi, A., Kabickova, E., and Woessmann, W.

Subjects
ANAPLASTIC large-cell lymphoma, CUTANEOUS T-cell lymphoma, ANAPLASTIC thyroid cancer
Abstract

We investigated all patients with ALK-positive ALCL with exclusive cytoplasmic ALK-expression diagnosed in a uniformly treated cohort of ALCL patients in the NHL-BFM study group. B Introduction: b ALK-positive anaplastic large cell lymphomas (ALCL) are characterized by rearrangements involving the I ALK i gene on chromosome 2. ALK-POSITIVE ANAPLASTIC LARGE CELL LYMPHOMA WITH VARIANT ALK-FUSION PARTNER: A POPULATION-BASED ANALYSES OF THE NHL-BFM STUDY GROUP. [Extracted from the article]

Published
2023
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21. LG-03 * RADIATION THERAPY IS ASSOCIATED WITH INCREASED LATE MORTALITY IN LONG-TERM ADULT SURVIVORS OF CHILDHOOD LOW GRADE GLIOMA: A POPULATION BASED STUDY

Author

Krishnatry, R., primary, Zhukova, N., additional, Stucklin, A. G., additional, Pole, J., additional, Mistry, M., additional, Ramaswamy, V., additional, Bartels, U., additional, Huang, A., additional, Laperriere, N., additional, Dirks, P., additional, Zelcer, S., additional, Silva, M., additional, Johnston, D. L., additional, Scheinemann, K., additional, Hawkins, C., additional, Bandopadhayay, P., additional, Kieran, M. W., additional, Manley, P. E., additional, Bouffet, E., additional, and Tabori, U., additional

Published
2015
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22. Genomic analysis of diffuse intrinsic pontine gliomas identifies three molecular subgroups and recurrent activating ACVR1 mutations.

Author

Dunn S., Cain J., Brennan C., Souweidane M.M., Jones C., Allis C.D., Brudno M., Becher O., Buczkowicz P., Hoeman C., Rakopoulos P., Pajovic S., Letourneau L., Dzamba M., Morrison A., Lewis P., Bouffet E., Bartels U., Zuccaro J., Agnihotri S., Ryall S., Barszczyk M., Chornenkyy Y., Bourgey M., Bourque G., Montpetit A., Cordero F., Castelo-Branco P., Mangerel J., Tabori U., Ho K.C., Huang A., Taylor K.R., Mackay A., Bendel A.E., Nazarian J., Fangusaro J.R., Karajannis M.A., Zagzag D., Foreman N.K., Donson A., Hegert J.V., Smith A., Chan J., Lafay-Cousin L., Hawkins C., Hukin J., Dunham C., Scheinemann K., Michaud J., Zelcer S., Ramsay D., Dunn S., Cain J., Brennan C., Souweidane M.M., Jones C., Allis C.D., Brudno M., Becher O., Buczkowicz P., Hoeman C., Rakopoulos P., Pajovic S., Letourneau L., Dzamba M., Morrison A., Lewis P., Bouffet E., Bartels U., Zuccaro J., Agnihotri S., Ryall S., Barszczyk M., Chornenkyy Y., Bourgey M., Bourque G., Montpetit A., Cordero F., Castelo-Branco P., Mangerel J., Tabori U., Ho K.C., Huang A., Taylor K.R., Mackay A., Bendel A.E., Nazarian J., Fangusaro J.R., Karajannis M.A., Zagzag D., Foreman N.K., Donson A., Hegert J.V., Smith A., Chan J., Lafay-Cousin L., Hawkins C., Hukin J., Dunham C., Scheinemann K., Michaud J., Zelcer S., and Ramsay D.

Abstract

Diffuse intrinsic pontine glioma (DIPG) is a fatal brain cancer that arises in the brainstem of children, with no effective treatment and near 100% fatality. The failure of most therapies can be attributed to the delicate location of these tumors and to the selection of therapies on the basis of assumptions that DIPGs are molecularly similar to adult disease. Recent studies have unraveled the unique genetic makeup of this brain cancer, with nearly 80% found to harbor a p.Lys27Met histone H3.3 or p.Lys27Met histone H3.1 alteration. However, DIPGs are still thought of as one disease, with limited understanding of the genetic drivers of these tumors. To understand what drives DIPGs, we integrated whole-genome sequencing with methylation, expression and copy number profiling, discovering that DIPGs comprise three molecularly distinct subgroups (H3-K27M, silent and MYCN) and uncovering a new recurrent activating mutation affecting the activin receptor gene ACVR1 in 20% of DIPGs. Mutations in ACVR1 were constitutively activating, leading to SMAD phosphorylation and increased expression of the downstream activin signaling targets ID1 and ID2. Our results highlight distinct molecular subgroups and novel therapeutic targets for this incurable pediatric cancer. © 2014 Nature America, Inc. All rights reserved.

Published
2014

24. RADIOLOGY

Author

Kelly, T., primary, Prah, M., additional, Jogal, S., additional, Maheshwari, M., additional, Lew, S., additional, Schmainda, K., additional, Kannan, G., additional, Khatua, S., additional, Zaky, W., additional, Ketonen, L., additional, Drogosiewicz, M., additional, Dembowska-Baginska, B., additional, Jurkiewicz, E., additional, Nowak, K., additional, Perek, D., additional, Hirpara, D., additional, Bhatt, M., additional, Scheinemann, K., additional, Shimizu, Y., additional, Kondo, A., additional, Miyajima, M., additional, Arai, H., additional, Dvir, R., additional, Shiran, S., additional, Sira, L. B.-, additional, Roth, J., additional, Tabori, U., additional, Bouffet, E., additional, Durno, C., additional, Aronson, M., additional, Constantini, S., additional, Elhasid, R., additional, Fangusaro, J., additional, Marsh, J., additional, Bregman, C., additional, Diaz, A., additional, Byrne, R., additional, Ziel, E., additional, Goldman, S., additional, Calmon, R., additional, Grevent, D., additional, Blauwblomme, T., additional, Puget, S., additional, Sainte-Rose, C., additional, Varlet, P., additional, Dufour, C., additional, Grill, J., additional, Saitovich, A., additional, Zilbovicius, M., additional, Brunelle, F., additional, Boddaert, N., additional, Wei, L., additional, Tan, A. M., additional, Tang, P. H., additional, Orphanidou-Vlachou, E., additional, Vlachos, N., additional, Davies, N., additional, Arvanitis, T., additional, Grundy, R., additional, Peet, A., additional, Withey, S., additional, Novak, J., additional, MacPherson, L., additional, Avula, S., additional, Kumar, R., additional, Pizer, B., additional, Pettorini, B., additional, Garlick, D., additional, Mallucci, C., additional, Reddick, W., additional, Guo, J., additional, Glass, J., additional, Pryweller, J., additional, Gajjar, A., additional, Thust, S., additional, Blanco, E., additional, Mankad, K., additional, and Michalski, A., additional

Published
2014
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25. ATYPICAL TERATOID RHABDOID TUMOUR

Author

Bertozzi, A. I., primary, Munzer, C., additional, Fouyssac, F., additional, Andre, N., additional, Boetto, S., additional, Leblond, P., additional, Bourdeaut, F., additional, Dufour, C., additional, Deshpande, R. K., additional, Bhat, K. G., additional, Mahalingam, S., additional, Muscat, A., additional, Cain, J., additional, Ferguson, M., additional, Popovski, D., additional, Algar, E., additional, Rossello, F. J., additional, Jayasekara, S., additional, Watkins, D. N., additional, Hodge, J., additional, Ashley, D., additional, Hishii, M., additional, Saito, M., additional, Arai, H., additional, Han, Z. Y., additional, Richer, W., additional, Lucchesi, C., additional, Freneaux, P., additional, Nicolas, A., additional, Grison, C., additional, Pierron, G., additional, Delattre, O., additional, Epari, S., additional, TS, N., additional, Gupta, T., additional, Chinnaswamy, G., additional, Sastri, J. G., additional, Shetty, P., additional, Moiyadi, A., additional, Jalali, R., additional, Fay-McClymont, T., additional, Johnston, D., additional, Janzen, L., additional, Guger, S., additional, Scheinemann, K., additional, Fleming, A., additional, Fryer, C., additional, Hukin, J., additional, Mabbott, D., additional, Huang, A., additional, Bouffet, E., additional, Lafay-Cousin, L., additional, Kawamura, A., additional, Yamamoto, K., additional, Nagashima, T., additional, Bartelheim, K., additional, Benesch, M., additional, Buchner, J., additional, Gerss, J., additional, Hasselblatt, M., additional, Kortmann, R.-D., additional, Fleischack, G., additional, Quiroga, E., additional, Reinhard, H., additional, Schneppenheim, R., additional, Seeringer, A., additional, Siebert, R., additional, Timmermann, B., additional, Warmuth-Metz, M., additional, Schmid, I., additional, Fruhwald, M. C., additional, Kerl, K., additional, Klingebiel, T., additional, Al-Kofide, A., additional, Khafaga, Y., additional, Al-Hindi, H., additional, Dababo, M., additional, Ul-Haq, A., additional, Anas, M., additional, Barria, M. G., additional, Siddiqui, K., additional, Hassounah, M., additional, Ayas, M., additional, Al-Shail, E., additional, Jeibmann, A., additional, Eikmeier, K., additional, Linge, A., additional, Johann, P., additional, Koos, B., additional, Kool, M., additional, Pfister, S. M., additional, Paulus, W., additional, Schuller, U., additional, Junckerstorff, R., additional, Rosenblum, M. K., additional, Alassiri, A. H., additional, Rossi, S., additional, Gottardo, N., additional, Toledano, H., additional, Viscardi, E., additional, Witkowski, L., additional, Nagel, I., additional, Oyen, F., additional, Foulkes, W. D., additional, Schrey, D., additional, Malietzis, G., additional, Chi, S., additional, Marshall, L., additional, Carceller, F., additional, Moreno, L., additional, Zacharoulis, S., additional, Bhardwaj, R., additional, Chakravadhanula, M., additional, Ozals, V., additional, Hampton, C., additional, Metpally, R., additional, Grillner, P., additional, Asmundsson, J., additional, Gustavsson, B., additional, Holm, S., additional, Johann, P. D., additional, Korshunov, A., additional, Ryzhova, M., additional, Milde, T., additional, Witt, O., additional, Jones, D. T. W., additional, Hovestadt, V., additional, Gajjar, A., additional, Fruhwald, M., additional, Pfister, S., additional, Finetti, M., additional, Pons, A. d. C., additional, Selby, M., additional, Smith, A., additional, Crosier, S., additional, Wood, J., additional, Skalkoyannis, B., additional, Bailey, S., additional, Clifford, S., additional, Williamson, D., additional, Rutkowski, S., additional, Kortmann, R. D., additional, Graf, N., additional, Boos, J., additional, Nysom, K., additional, Moreno, N., additional, Holsten, T., additional, Ahlfeld, J., additional, Mertins, J., additional, Hotfilder, M., additional, Schleicher, S., additional, Handgretinger, R., additional, Meisterernst, M., additional, Schmidt, C., additional, Dittmar, S., additional, Chan, G. C. F., additional, Shing, M. M. K., additional, Yuen, H. L., additional, Li, R. C. H., additional, Ling, S. L., additional, Slavc, I., additional, Peyrl, A., additional, Chocholous, M., additional, Azizi, A., additional, Czech, T., additional, Dieckmann, K., additional, Haberler, C., additional, Leiss, U., additional, Gotti, G., additional, Biassoni, V., additional, Schiavello, E., additional, Spreafico, F., additional, Pecori, E., additional, Gandola, L., additional, Massimino, M., additional, Kornelius, K., additional, Yano, H., additional, Nakayama, N., additional, Ohe, N., additional, Ozeki, M., additional, Kanda, K., additional, Kimura, T., additional, Hori, T., additional, Fukao, T., additional, Iwama, T., additional, Weil, A. G., additional, Diaz, A., additional, Gernsback, J., additional, Bhatia, S., additional, Ragheb, J., additional, Niazi, T., additional, Khatib, Z., additional, Zoghbi, A., additional, Meisterernst, a. M., additional, Birks, D., additional, Griesinger, A., additional, Amani, V., additional, Donson, A., additional, Posner, R., additional, Dunham, C., additional, Kleinschmidt-DeMasters, B. K., additional, Handler, M., additional, Vibhakar, R., additional, Foreman, N., additional, Zhou, L., additional, Catchpoole, D., additional, Kakkar, A., additional, Biswas, A., additional, Suri, V., additional, Sharma, M., additional, Kale, S., additional, Mahapatra, A., additional, Sarkar, C., additional, Torchia, J., additional, Picard, D., additional, Ho, K. C., additional, Khuong-Quang, D.-A., additional, Louterneau, L., additional, Bourgey, M., additional, Chan, T., additional, Golbourn, B., additional, Cousin, L.-L., additional, Taylor, M. D., additional, Dirks, P., additional, Rutka, J. T., additional, Hawkins, C., additional, Majewski, J., additional, Kim, S.-K., additional, Jabado, N., additional, Chang, J. H.-C., additional, Confer, M., additional, Chang, A., additional, Goldman, S., additional, Dunn, M., additional, and Hartsell, W., additional

Published
2014
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26. PEDIATRICS CLINICAL RESEARCH

Author

Antony, R., primary, Zagardo, M., additional, Gujrati, M., additional, Lin, J., additional, Antony, R., additional, Al-Rahawan, M., additional, Broniscer, A., additional, Bhardwaj, R., additional, Hampton, C., additional, Ozols, V., additional, Chakravadhanula, M., additional, Bouffet, E., additional, Hawkins, C., additional, Scheinemann, K., additional, Zelcer, S., additional, Johnston, D., additional, Lafay-Cousin, L., additional, Larouche, V., additional, Jabado, N., additional, Carret, A. S., additional, Hukin, J., additional, Eisenstat, D., additional, Pond, G., additional, Poskitt, K., additional, Wilson, B., additional, Bartels, U., additional, Tabori, U., additional, Dhall, G., additional, Haley, K., additional, Finlay, J., additional, Rushing, T., additional, Sposto, R., additional, Seeger, R., additional, Garvin, J., additional, Rupani, K., additional, Stark, E., additional, Anderson, R., additional, Feldstein, N., additional, Grill, J., additional, Hargrave, D., additional, Massimino, M., additional, Jaspan, T., additional, Varlet, P., additional, Jones, C., additional, Morgan, P., additional, Le Deley, M. C., additional, Azizi, A., additional, Canete, A., additional, Saran, F., additional, Bachir, J., additional, Bubuteishvili-Pacaud, L., additional, Rousseau, R., additional, Vassal, G., additional, Gupta, S., additional, Robinson, N., additional, Dhir, N., additional, Wong, K., additional, Zhou, S., additional, Kumabe, T., additional, Kawaguchi, T., additional, Saito, R., additional, Kanamori, M., additional, Yamashita, Y., additional, Sonoda, Y., additional, Tominaga, T., additional, Miyagawa, T., additional, Nwachukwu, C., additional, Youland, R., additional, Laack, N., additional, Filipek, I., additional, Drogosiewicz, M., additional, Polnik, M. P.-, additional, Swieszkowska, E., additional, Dembowska-Baginska, B., additional, Jurkiewicz, E., additional, Perek, D., additional, Grajkowska, W., additional, Roszkowski, M., additional, Sobol, G., additional, Musiol, K., additional, Wachowiak, J., additional, Kazmierczak, B., additional, Pogorzelski, J. P. -, additional, Mlynarski, W., additional, Szewczyk, B. Z.-, additional, Wysocki, M., additional, Niedzielska, E., additional, Kowalczyk, J., additional, Slusarz, H. W. -, additional, Balwierz, W., additional, Czepko, E. Z. -, additional, Szolkiewicz, A., additional, Perek-Polnik, M., additional, Lastowska, M., additional, Chojnacka, M., additional, Tarasinska, M., additional, Perreault, S., additional, Chao, K., additional, Ramaswamy, V., additional, Shih, D., additional, Remke, M., additional, Luu, B., additional, Schubert, S., additional, Fisher, P., additional, Partap, S., additional, Vogel, H., additional, Taylor, M., additional, Goumnerova, L., additional, Cho, Y.-J., additional, Robison, N., additional, Brown, R., additional, Cloughesy, T., additional, Davidson, T. B., additional, Krieger, M., additional, Berger, M., additional, Perry, A., additional, Gilles, F., additional, Finlay, J. L., additional, Khemani, J., additional, Britt, B., additional, Grimm, J., additional, Ruge, M. I., additional, Blau, T., additional, Hafkemeyer, V., additional, Hamisch, C., additional, Klinger, K., additional, Simon, T., additional, Sadighi, Z., additional, Ellezam, B., additional, Guindani, M., additional, Ater, J., additional, Shimizu, Y., additional, Arai, H., additional, Miyajima, M., additional, Shimoji, K., additional, Kondo, A., additional, Shinohara, E., additional, Perkins, S., additional, DeWees, T., additional, Slavc, I., additional, Chocholous, M., additional, Leiss, U., additional, Haberler, C., additional, Peyrl, A., additional, Azizi, A. A., additional, Dieckmann, K., additional, Woehrer, A., additional, Dorfer, C., additional, Czech, T., additional, Spence, T., additional, Picard, D., additional, Barszczyk, M., additional, Kim, S.-K., additional, Ra, Y.-S., additional, Fangusaro, J., additional, Toledano, H., additional, Nakamura, H., additional, Fan, X., additional, Muraszko, K. M., additional, Ng, H.-K., additional, Halliday, W., additional, Shago, M., additional, Hawkins, C. E., additional, Huang, A., additional, Suzuki, M., additional, van Zanten, S. V., additional, Jansen, M., additional, van Vuurden, D., additional, Hulleman, E., additional, Idema, S., additional, Noske, D., additional, Wolf, N., additional, Hendrikse, H., additional, Vandertop, P., additional, Kaspers, G. J., additional, Muller, K., additional, Schlamann, A., additional, Warmuth-Metz, M., additional, Pietsch, T., additional, Pietschmann, S., additional, Kortmann, R.-D., additional, Kramm, C. M., additional, von Bueren, A. O., additional, Walston, S., additional, Williams, T., additional, Hamstra, D., additional, Oh, K., additional, Pelloski, C., additional, Zhukova, N., additional, Pole, J., additional, Mistry, M., additional, Fried, I., additional, Lapperiere, N., additional, Dirks, P., additional, An, J., additional, Alon, N., additional, Nathan, P., additional, Greenberg, M., additional, and Malkin, D., additional

Published
2013
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28. QUALITY OF LIFE/AFTERCARE

Author

Rednam, S., primary, Scheurer, M., additional, Adesina, A., additional, Lau, C., additional, Okcu, M., additional, Deatrick, J., additional, Ogle, S., additional, Fisher, M., additional, Barakat, L., additional, Hardie, T., additional, Li, Y., additional, Ginsberg, J., additional, Ben-Arush, M., additional, Krivoy, E., additional, Rosenkranz, R., additional, Peretz-Nahum, M., additional, Brown, R. J., additional, Love, J., additional, Warburton, D., additional, McBride, W. H., additional, Bluml, S., additional, Mueller, S., additional, Sear, K., additional, Hills, N., additional, Chettout, N., additional, Afghani, S., additional, Lew, L., additional, Tolentino, E., additional, Haas-Kogan, D., additional, Fullerton, H., additional, Reddick, W., additional, Palmer, S., additional, Glass, J., additional, Ogg, R., additional, Gajjar, A., additional, Omar, A., additional, Perkins, S., additional, Shinohara, E., additional, Spoljaric, D., additional, Isenberg, J., additional, Whittington, M., additional, Hauff, M., additional, King, A., additional, Litzelman, K., additional, Barker, E., additional, Catrine, K., additional, Puccetti, D., additional, Possin, P., additional, Witt, W., additional, Mallucci, C., additional, Kumar, R., additional, Pizer, B., additional, Williams, D., additional, Pettorini, B., additional, Piscione, J., additional, Bouffet, E., additional, Shams, I., additional, Kulkarni, A., additional, Remes, T., additional, Harila-Saari, A., additional, Suo-Palosaari, M., additional, Arikoski, P., additional, Riikonen, P., additional, Sutela, A., additional, Koskenkorva, P., additional, Ojaniemi, M., additional, Rantala, H., additional, Campen, C. J., additional, Ashby, D., additional, Fisher, P. G., additional, Monje, M., additional, Kulkarni, A. V., additional, Nakamura, H., additional, Makino, K., additional, Yano, S., additional, Kuratsu, J.-i., additional, Jadrijevic-Cvrlje, F., additional, Batinica, M., additional, Toledano, H., additional, Hoffman, T., additional, Ezer-Cohen, Y., additional, Michowiz, S., additional, Yaniv, I., additional, Cohen, I. J., additional, Adler, I., additional, Mindel, S., additional, Gopalakrishnamoorthy, M., additional, Saunders, D., additional, Gaze, M., additional, Spoudeas, H., additional, Kieffer, V., additional, Dellatolas, G., additional, Chevignard, M., additional, Puget, S., additional, Dhermain, F., additional, Grill, J., additional, Dufour, C., additional, Muir, R., additional, Hunter, A., additional, Latchman, A., additional, de Camargo, O., additional, Scheinemann, K., additional, Dhir, N., additional, Zaky, W., additional, Zomorodian, T., additional, Wong, K., additional, Dhall, G., additional, Macy, M., additional, Lauro, C., additional, Zeitler, P., additional, Foreman, N., additional, Liu, A., additional, Chocholous, M., additional, Dodier, P., additional, Peyrl, A., additional, Dieckmann, K., additional, Hausler, G., additional, Slavc, I., additional, Avula, S., additional, Garlick, D., additional, Armstrong, G., additional, Kawashima, T., additional, Leisenring, W., additional, Stovall, M., additional, Sklar, C., additional, Robison, L., additional, Samaan, C., additional, Duckworth, J., additional, Greenberg-Kushnir, N., additional, Freedman, S., additional, Eshel, R., additional, Zverling, N., additional, Elhasid, R., additional, Dvir, R., additional, Yalon, M., additional, Constantini, S., additional, Wilne, S., additional, Liu, J.-F., additional, Trusler, J., additional, Lundsell, S., additional, Kennedy, C., additional, Clough, L., additional, Dickson, N., additional, Lakhanpaul, M., additional, Baker, M., additional, Dudley, J., additional, Grundy, R., additional, Walker, D., additional, von Hoff, K., additional, Herzog, N., additional, Ottensmeier, H., additional, Grabow, D., additional, Gerber, N. U., additional, Friedrich, C., additional, von Bueren, A. O., additional, Resch, A., additional, Kortmann, R. D., additional, Kaatsch, P., additional, Doerr, H. G., additional, Rutkowski, S., additional, del Bufalo, F., additional, Mastronuzzi, A., additional, Serra, A., additional, de Sio, L., additional, Locatelli, F., additional, Biassoni, V., additional, Leonardi, M., additional, Ajovalasit, D., additional, Riva, D., additional, Vago, C., additional, Usilla, A., additional, Fidani, P., additional, Schiavello, E., additional, Gariboldi, F., additional, Massimino, M., additional, Lober, R., additional, Perrault, S., additional, Partap, S., additional, Edwards, M., additional, Fisher, P., additional, Yeom, K., additional, Salgado, D., additional, Nunes, S., additional, Vinhais, S., additional, Wells, E. M., additional, Seidel, K., additional, Ullrich, N. J., additional, Diller, L., additional, Krull, K. R., additional, Neglia, J., additional, Robison, L. L., additional, Whelan, K., additional, Russell, C. E., additional, Brownstone, D., additional, Kaise, C., additional, Bull, K., additional, Culliford, D., additional, Calaminus, G., additional, Bertin, D., additional, Vallero, S., additional, Romano, E., additional, Basso, M. E., additional, Biasin, E., additional, Fagioli, F., additional, Ziara, K., additional, L'Hotta, A., additional, Williams, A., additional, Thede, R., additional, Moore, K., additional, James, A., additional, Bjorn, E., additional, Franzen, P., additional, Haag, A., additional, Lax, A.-K., additional, Moreno, I., additional, Obeid, J., additional, Timmons, B. W., additional, Iwata, W., additional, Wagner, S., additional, Lai, J.-S., additional, Waddell, K., additional, VanLeeuwen, S., additional, Newmark, M., additional, Noonan, J., additional, O'Connell, K., additional, Urban, M., additional, Yount, S., additional, Goldman, S., additional, Igoe, D., additional, Cunningham, T., additional, Orfus, M., additional, Mabbott, D., additional, Liptak, C., additional, Manley, P., additional, Recklitis, C., additional, Zhang, P., additional, Shaikh, F., additional, Narang, I., additional, Matsumoto, K., additional, Yamasaki, K., additional, Okada, K., additional, Fujisaki, H., additional, Osugi, Y., additional, Hara, J., additional, Phipps, K., additional, Gumley, D., additional, Jacques, T., additional, Hargrave, D., additional, Michalski, A., additional, Chordas, C., additional, Chi, S., additional, Robison, N., additional, Bandopadhayay, P., additional, Marcus, K., additional, Zimmerman, M. A., additional, Goumnerova, L., additional, Kieran, M., additional, Brand, S., additional, Brinkman, T., additional, Delaney, B., additional, Diver, T., additional, Rey, C., additional, Madden, J. R., additional, Hemenway, M. S., additional, Dorneman, L., additional, Stiller, D., additional, Liu, A. K., additional, Foreman, N. K., additional, Vibhakar, R., additional, Mitchell, M., additional, Hemenway, M., additional, Madden, J., additional, Ryan, M., additional, O'Kane, R., additional, Picton, S., additional, Kenny, T., additional, Stiller, C., additional, Chumas, P., additional, Bendel, A., additional, Patterson, R., additional, Barrera, M., additional, Schulte, F., additional, Bartels, U., additional, Janzen, L., additional, Johnston, D., additional, Cataudella, D., additional, Chung, J., additional, Sung, L., additional, Hancock, K., additional, Hukin, J., additional, Zelcer, S., additional, Brandon, S., additional, Montour-Proulx, I., additional, Strother, D., additional, Cooksey, R., additional, Bowers, D., additional, Gargan, L., additional, Gode, A., additional, Klesse, L., additional, Oden, J., additional, Vega, G., additional, Sala, F., additional, Nuzzi, D., additional, Mulino, M., additional, Masotto, B., additional, Mazza, C., additional, Bricolo, A., additional, Gerosa, M., additional, Tong, M., additional, Laughlin, S., additional, Mackie, S., additional, Taylor, L., additional, Sharpe, G., additional, Al-Salihi, O., additional, and Nicolin, G., additional

Published
2012
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29. EPIDEMIOLOGY

Author

Khatua, S., primary, Brown, R., additional, Pearlman, M., additional, Vats, T., additional, Satge, D., additional, Stiller, C., additional, Rutkowski, S., additional, von Bueren, A. O., additional, Lacour, B., additional, Sommelet, D., additional, Nishi, M., additional, Massimino, M., additional, Garre, M.-L., additional, Moreno, F., additional, Hasle, H., additional, Jakab, Z., additional, Greenberg, M., additional, von der Weid, N., additional, Kuehni, C., additional, Zurriaga, O., additional, Vicente, M.-L., additional, Peris-Bonet, R., additional, Benesch, M., additional, Vekemans, M., additional, Sullivan, S., additional, Rickert, C., additional, Fisher, P. G., additional, Von Behren, J., additional, Nelson, D. O., additional, Reynolds, P., additional, Fukuoka, K., additional, Yanagisawa, T., additional, Suzuki, T., additional, Koga, T., additional, Wakiya, K., additional, Adachi, J.-i., additional, Mishima, K., additional, Fujimaki, T., additional, Matsutani, M., additional, Nishikawa, R., additional, Gidding, C., additional, Schieving, J., additional, Wesseling, P., additional, Ligtenberg, M., additional, Hoogerbrugge, N., additional, Jongmans, M., additional, Crosier, S., additional, Nicholson, S. L., additional, Robson, K., additional, Jacques, T., additional, Wharton, S., additional, Bown, N., additional, Michalski, A., additional, Pizer, B., additional, Clifford, S., additional, Sanden, E., additional, Visse, E., additional, Siesjo, P., additional, Darabi, A., additional, Nousome, D., additional, Lupo, P. J., additional, Scheurer, M. E., additional, Nulman, I., additional, Barrera, M., additional, Maxwell, C., additional, Koren, G., additional, Gorelyshev, S., additional, Matuev, K., additional, Lubnin, A., additional, Laskov, M., additional, Lemeneva, N., additional, Mazerkina, N., additional, Khuhlaeva, E., additional, Muller, K., additional, Bruns, F., additional, Pietsch, T., additional, Kortmann, R.-D., additional, Krishnatry, R., additional, Shirsat, N., additional, Kunder, R., additional, Epari, S., additional, Gupta, T., additional, Kurkure, P., additional, Vora, T., additional, Arora, B., additional, Moiyadi, A., additional, Jalali, R., additional, Swieszkowska, E., additional, Dembowska-Baginska, B., additional, Drogosiewicz, M., additional, Filipek, I., additional, Perek-Polnik, M., additional, Grajkowska, W., additional, Perek, D., additional, Johnston, D., additional, Cyr, J., additional, Strother, D., additional, Lafay-Cousin, L., additional, Fryer, C., additional, Scheinemann, K., additional, Carret, A.-S., additional, Fleming, A., additional, Larouche, V., additional, Bouffet, E., additional, Friedrich, C., additional, Gnekow, A. K., additional, Fleischhack, G., additional, Kramm, C. M., additional, Fruehwald, M. C., additional, Muller, H. L., additional, Calaminus, G., additional, Kordes, U., additional, Faldum, A., additional, Warmuth-Metz, M., additional, Kortmann, R. D., additional, Jung, I., additional, Kaatsch, P., additional, Caretti, V., additional, Bugiani, M., additional, Boor, I., additional, Schellen, P., additional, Vandertop, W. P., additional, Noske, D. P., additional, Kaspers, G., additional, Wurdinger, T., additional, Robinson, G., additional, Chingtagumpala, M., additional, Adesina, A., additional, Dalton, J., additional, Santi, M., additional, Sievert, A., additional, Wright, K., additional, Armstrong, G., additional, Boue, D., additional, Olshefski, R., additional, Scott, S., additional, Huang, A., additional, Cohn, R., additional, Gururangan, S., additional, Bowers, D., additional, Gilbertson, R., additional, Gajjar, A., additional, Ellison, D., additional, Chick, E., additional, Donson, A., additional, Owens, E., additional, Smith, A. A., additional, Madden, J. R., additional, Foreman, N. K., additional, Bakry, D., additional, Aronson, M., additional, Durno, C., additional, Hala, R., additional, Farah, R., additional, Amayiri, N., additional, Alharbi, Q., additional, Shamvil, A., additional, Ben-Shachar, S., additional, Constantini, S., additional, Rina, D., additional, Ellise, J., additional, Keiles, S., additional, Pollet, A., additional, Qaddoumi, I., additional, Gallinger, S., additional, Malkin, D., additional, Hawkins, C., additional, Tabori, U., additional, Trivedi, M., additional, Goodden, J., additional, Chumas, P., additional, Tyagi, A., additional, O'kane, R., additional, O'Kane, R., additional, Crimmins, D., additional, Picton, S., additional, and Elliott, M., additional

Published
2012
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30. RADIOLOGY

Author

Murray, J., primary, Braly, E., additional, Head, H., additional, Donahue, D., additional, Rush, S., additional, Stence, N., additional, Liu, A., additional, Kleinhenz, J., additional, Bison, B., additional, Pietsch, T., additional, von Hoff, K., additional, von Bueren, A., additional, Rutkowski, S., additional, Warmuth-Metz, M., additional, Jaspan, T., additional, Brisse, H., additional, Potepan, P., additional, Berg, F., additional, Gerber, N., additional, Sugiyama, K., additional, Kurisu, K., additional, Kajiwara, Y., additional, Takayasu, T., additional, Saito, T., additional, Hanaya, R., additional, Yamasaki, F., additional, Vicente, J., additional, Fuster-Garcia, E., additional, Tortajada, S., additional, Garcia-Gomez, J. M., additional, Davies, N., additional, Natarajan, K., additional, Wilson, M., additional, Grundy, R. G., additional, Wesseling, P., additional, Monleon, D., additional, Celda, B., additional, Robles, M., additional, Peet, A. C., additional, Perret, C., additional, Boltshauser, E., additional, Scheer, I., additional, Kellenberger, C., additional, Grotzer, M., additional, Steffen-Smith, E., additional, Venzon, D., additional, Bent, R., additional, Baker, E., additional, Shandilya, S., additional, Warren, K., additional, Shih, C.-S., additional, West, J., additional, Ho, C., additional, Porter, D., additional, Wang, Y., additional, Saykin, A., additional, McDonald, B., additional, Arfanakis, K., additional, Vezina, G., additional, Hargrave, D., additional, Poussaint, T. Y., additional, Goldman, S., additional, Packer, R., additional, Wen, P., additional, Pollack, I., additional, Zurakowski, D., additional, Kun, L., additional, Prados, M., additional, Kieran, M., additional, Eckel, L., additional, Keating, G., additional, Giannini, C., additional, Wetjen, N., additional, Patton, A., additional, Sarlls, J., additional, Pierpaoli, C., additional, Walker, L., additional, Perreault, S., additional, Lober, R., additional, Yeom, K., additional, Carret, A.-S., additional, Vogel, H., additional, Partap, S., additional, Fisher, P., additional, Gill, S. K., additional, Davies, N. P., additional, MacPherson, L., additional, Arvanitis, T. N., additional, Gill, S., additional, Arvanitis, T., additional, Peet, A., additional, Hayes, L., additional, Jones, R., additional, Mazewski, C., additional, Aguilera, D., additional, Palasis, S., additional, Bendel, A., additional, Patterson, R., additional, Petronio, J., additional, Meijer, L., additional, Grundy, R. G. G., additional, Walker, D. A., additional, Robison, N., additional, Grant, F., additional, Treves, S. T., additional, Bandopadhayay, P., additional, Manley, P., additional, Chi, S., additional, Zimmerman, M. A., additional, Chordas, C., additional, Goumnerova, L., additional, Smith, E., additional, Scott, M., additional, Ullrich, N. J., additional, Poussaint, T., additional, Yang, J. C., additional, Lightner, D. D., additional, Khakoo, Y., additional, Wolden, S. L., additional, Smee, R., additional, Zhao, C., additional, Spencer-Trotter, B., additional, Hallock, A., additional, Konski, A., additional, Bhambani, K., additional, Mahajan, A., additional, Jones, J., additional, Ketonen, L., additional, Paulino, A., additional, Ater, J., additional, Grosshans, D., additional, Dauser, R., additional, Weinberg, J., additional, Chintagumpala, M., additional, Dvir, R., additional, Elhasid, R., additional, Corn, B., additional, Tempelhoff, H., additional, Matceyevsky, D., additional, Makrin, V., additional, Shtraus, N., additional, Yavetz, D., additional, Constantini, S., additional, Gez, E., additional, Yu, E.-S., additional, Kim, Y.-J., additional, Park, H. J., additional, Kim, H. J., additional, Shin, S. H., additional, Kim, J.-H., additional, Kim, J.-Y., additional, Lee, Y. K., additional, Fiore, M. R., additional, Sanne, C., additional, Mandeville, H. C., additional, Saran, F. H., additional, Greenspoon, J., additional, Duckworth, J., additional, Singh, S., additional, Scheinemann, K., additional, Whitton, A., additional, Gauvain, K., additional, Geller, T., additional, Elbabaa, S., additional, Dombrowski, J., additional, Wong, K., additional, Olch, A., additional, Davidson, T. B., additional, Venkatramani, R., additional, Haley, K., additional, Zaky, W., additional, Dhall, G., additional, Finlay, J., additional, Bishop, M. W., additional, Hummel, T. R., additional, Leach, J., additional, Minturn, J., additional, Breneman, J., additional, Stevenson, C., additional, Wagner, L., additional, Sutton, M., additional, Miles, L., additional, and Fouladi, M., additional

Published
2012
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31. GERM CELL TUMORS

Author

Yang, Q.-y., primary, Chen, Z.-p., additional, Hayase, T., additional, Gomi, A., additional, Higaki, A., additional, Kawahara, Y., additional, Kobari, T., additional, Fukuda, T., additional, Kashii, Y., additional, Morimoto, A., additional, Sakatani, T., additional, Momoi, M. Y., additional, Murray, M., additional, Hale, J., additional, Heinemann, K., additional, Saran, F., additional, Calaminus, G., additional, Nicholson, J., additional, Martinez, S., additional, Khakoo, Y., additional, Gilheeney, S., additional, Kramer, K., additional, Wolden, S., additional, Souweidane, M., additional, Dunkel, I., additional, Brichtova, E., additional, Pavelka, Z., additional, Bobekova, A., additional, Magnova, O., additional, Kren, L., additional, Svoboda, T., additional, Sprlakova, A., additional, Slampa, P., additional, Zitterbart, K., additional, Sterba, J., additional, Campen, C. J., additional, Ashby, D., additional, Fisher, P. G., additional, Monje, M., additional, Dagri, J., additional, Torkildson, J., additional, Cheng, J., additional, Wang, R. X., additional, Yock, T., additional, Banerjee, A., additional, Dhall, G., additional, Finlay, J., additional, Yanagisawa, T., additional, Fukuoka, K., additional, Suzuki, T., additional, Kohga, T., additional, Wakiya, K., additional, Adachi, J., additional, Mishima, K., additional, Fujimaki, T., additional, Matsutani, M., additional, Nishikawa, R., additional, Frappaz, D., additional, Kortmann, R. D., additional, Alapetite, C., additional, Garre, M. L., additional, Ricardi, U., additional, Saran, F. H., additional, Czech, T., additional, Walker, R., additional, Koga, T., additional, Legault, G., additional, Allen, J., additional, Geludkova, O., additional, Mushinskaya, M., additional, Kushel, Y., additional, Korshunov, A., additional, Melikyan, A., additional, Shishkina, L., additional, Oserova, V., additional, Oserov, S., additional, Maserkina, N., additional, Borodina, I., additional, Kumirova, E., additional, Boyarchuk, N., additional, Gorbatyh, S., additional, Popova, E., additional, Sherbenko, O., additional, Zelinskaya, N., additional, Shammasov, R., additional, Privalova, L., additional, Chulkov, O., additional, Kosel, Y., additional, Cappellano, A. M., additional, Paiva, P., additional, Cavalheiro, S., additional, Dastoli, P., additional, Seixas, M. T., additional, Silva, N. S., additional, Chan, G. C.-F., additional, Shing, M. M.-K., additional, Yuen, H.-L., additional, Li, R. C.-H., additional, Li, C.-K., additional, Ha, S.-Y., additional, Chen, H.-H., additional, Chang, F.-C., additional, Chen, Y.-W., additional, Wong, T.-T., additional, Yarascavitch, B., additional, Stein, N., additional, Ribeiro, L., additional, Whitton, A., additional, Duckworth, J., additional, Scheinemann, K., additional, Singh, S., additional, Ozerov, S., additional, Gorelyshev, S., additional, Trunin, Y., additional, Kagawa, N., additional, Fujimoto, Y., additional, Hirayama, R., additional, Chiba, Y., additional, Kijima, N., additional, Arita, H., additional, Kinoshita, M., additional, Hashimoto, N., additional, Maruno, M., additional, Yoshimine, T., additional, Guerra, G. P., additional, Oscanoa, M., additional, Cavero, L., additional, Yabar, A., additional, Ugarte, E., additional, Trivedi, M., additional, Tyagi, A., additional, Goodden, J., additional, Chumas, P., additional, Elliott, M., additional, Picton, S., additional, Robison, N., additional, Prabhu, S., additional, Sun, P., additional, Chi, S., additional, Kieran, M., additional, Manley, P., additional, Cohen, L., additional, Goumnerova, L., additional, Smith, E., additional, Scott, M., additional, London, W., additional, and Ullrich, N. J., additional

Published
2012
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35. Utility of peripheral blood cultures in bacteremic pediatric cancer patients with a central line.

Author

Scheinemann K, Ethier MC, Dupuis LL, Richardson SE, Doyle J, Allen U, Sung L, Scheinemann, Katrin, Ethier, Marie-Chantal, Dupuis, L Lee, Richardson, Susan E, Doyle, John, Allen, Upton, and Sung, Lillian

Abstract

Purpose: The utility of peripheral blood cultures in febrile neutropenic children with cancer and central venous catheters (CVC) is controversial. Our primary objective was to describe true bloodstream infections detected only by peripheral culture. Our secondary objectives were to describe true bloodstream infections detected only by CVC culture and to describe probable contaminants detected in both types of blood cultures.Methods: We included children with cancer who had peripheral and CVC cultures obtained on the same day in which at least one culture was positive. Only cultures obtained prior to the initiation of broad-spectrum antibiotics were included. We defined true bloodstream infections due to common contaminants (such as coagulase-negative Staphylococcus) as occurring if multiple cultures were positive for the same organism or if sepsis was present.Results: Between January 2002 and July 2007, 318 episodes of bloodstream infection from 224 children were included. Of these, 228/318 (71.7%) were classified as true bloodstream infections while 90/318 (28.3%) were classified as contaminants. Importantly, 28/228 (12.3%) true bloodstream infections were detected only in peripheral culture while 85/228 (37.3%) true bloodstream infections were detected only by CVC cultures. Contaminants were identified in peripheral culture in 45/318 (14.2%) of episodes and in CVC culture in 45/318 (14.2%) episodes.Conclusions: True bloodstream infections frequently are only detected in the peripheral culture. These data support continuation of the practice of routine peripheral cultures in addition to CVC cultures at the onset of fever for children with cancer who are not already receiving broad-spectrum antibiotics. [ABSTRACT FROM AUTHOR]

Published
2010
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37. Essential medicines for childhood cancer in Europe: a pan-European, systematic analysis by SIOPE

Author

Maria Otth, Eva Brack, Pamela R Kearns, Olga Kozhaeva, Marko Ocokoljic, Reineke A Schoot, Gilles Vassal, Federica Achini, Adriana Balduzzi, Maja Beck Popovic, Auke Beishuizen, Luca Bergamaschi, Andrea Biondi, Franck Bourdeaut, Elena Braicu, Jesper Brok, Laurence Brugières, Amos Burke, Gabriele Calaminus, Michela Casanova, Marie-Louise Choucair, Morgane Cleirec, Selim Corbaciouglu, Maria Genoveva Correa Llano, Teresa De Rojas, Nerea Domínguez Pinilla, Caroline Elmaraghi, Andrea Ferrari, Alexander Fossa, Nathalie Gaspar, Nikolas Herold, Kyriaki Karapiperi, Maarja Karu, Mimi Kjærsgaar, Fabian Knörr, Christa Koenig, Izabela Kranjcec, Malgorzata Krawczyk, Kai Lehmberg, Thomas Lehrnbecher, Maaike Lunesink, Davide Massano, Nuša Matijasic, Hans Merks, Markus Metzler, Anthony Michalski, Milen Minkov, Bruce Morland, Naghmeh Niktoreh, Elena Oltenau, Daniel Orbach, Cormac Owens, Smaragda Papachristidou, Claudia Pasqualini, Maja Pavlovic, Paula Perez Albert, Fiona Poyer, Ivana Radulovic, Dirk Reinhardt, Joana Rebelo, Eva Roser, Ida Russo, Katrin Scheinemann, Christina Schindera, Martin Schrappe, Astrid Sehested, Jalid Sehouli, Filippo Spreafico, Sandra J Strauss, Janine Stutterheim, Karel Svojgr, Vasiliki Tzotzola, Roelof Van Ewijk, Arnauld Verschuur, Ajay Vora, Willi Woessmann, Olga Zajac-Spychala, Michel Zwaan, Maria, O, Eva, B, Pamela R, K, Olga, K, Marko, O, Reineke A, S, Gilles, V, Achini, F, Balduzzi, A, Beck Popovic, M, Beishuizen, A, Bergamaschi, L, Biondi, A, Bourdeaut, F, Braicu, E, Brok, J, Brugières, L, Burke, A, Calaminus, G, Casanova, M, Choucair, M, Cleirec, M, Corbaciouglu, S, Genoveva Correa Llano, M, De Rojas, T, Domínguez Pinilla, N, Elmaraghi, C, Ferrari, A, Fossa, A, Gaspar, N, Herold, N, Karapiperi, K, Karu, M, Kjærsgaar, M, Knörr, F, Koenig, C, Kranjcec, I, Krawczyk, M, Lehmberg, K, Lehrnbecher, T, Lunesink, M, Massano, D, Matijasic, N, Merks, H, Metzler, M, Michalski, A, Minkov, M, Morland, B, Niktoreh, N, Oltenau, E, Orbach, D, Owens, C, Papachristidou, S, Pasqualini, C, Pavlovic, M, Perez Albert, P, Poyer, F, Radulovic, I, Reinhardt, D, Rebelo, J, Roser, E, Russo, I, Scheinemann, K, Schindera, C, Schrappe, M, Sehested, A, Sehouli, J, Spreafico, F, J Strauss, S, Stutterheim, J, Svojgr, K, Tzotzola, V, Van Ewijk, R, Verschuur, A, Vora, A, Woessmann, W, Zajac-Spychala, O, and Zwaan, M

Subjects
Europe, Adolescent, Oncology, Neoplasms, Childhood cancer, essential medicines, SIOPe, Humans, Antineoplastic Agents, Child, Medical Oncology, Drugs, Essential
Abstract

Background: Shortages and unequal access to anticancer medicines for children and adolescents are a reality in Europe. The aim of the European Society for Paediatric Oncology (SIOPE) Essential Anticancer Medicines Project was to provide a list of anticancer medicines that are considered essential in the treatment of paediatric cancers to help ensure their continuous access to all children and adolescents with cancer across Europe. Methods: This pan-European project, done between Jan 20, 2020, and Feb 18, 2022, was designed to be a systematic collection and review of treatment protocols and strategies that are used to treat childhood cancer in Europe. We formed 16 working groups on the basis of paediatric cancer types, and which were based on the existing SIOPE Clinical Trial Groups. Workings groups consisted of representatives from the SIOPE Clinical Trial Groups, Young SIOPE members, and senior paediatric oncology experts. Each group collected existing treatment protocols that are used to treat the respective cancer types in Europe. Medicines from the standard group of each protocol were extracted. For medicines not on the WHO Essential Medicines List for children (EMLc) 2017, working groups did a literature search to determine whether the medicines should be defined as essential, promising, or neither essential nor promising. Each group provided an individual summary, and all medicines that were considered essential by at least one group were combined in a joint list. Findings: The working groups identified 73 treatment protocols used in Europe and defined 66 medicines as essential. For several newer medicines, such as kinase inhibitors or tisagenlecleucel, the supporting evidence was insufficient to consider them essential, so these medicines were defined as promising. 25 medicines were considered promising by at least one working group. 22 (33%) of the 66 essential and none of the promising medicines were included in the WHO EMLc 2017. The WHO EMLc 2021 included two new medicines (everolimus and vinorelbine) following applications we made as a result of this project. Interpretation: Medicines that were defined as essential within this project should be available for the treatment of childhood and adolescent cancer continuously and across Europe. This list can be used to support and guide stakeholders and policy makers in negotiations on a national and European level regarding shortages, accessibility, and affordability of these medicines. Funding: None.

Published
2022
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38. Integrated (epi)-Genomic Analyses Identify Subgroup-Specific Therapeutic Targets in CNS Rhabdoid Tumors

Author

Constanze Zeller, Joseph D. Norman, Man Yu, Jian Qiang Lu, Doug Strother, Miklós Garami, C. Jane McGlade, Seung-Ki Kim, Misko Dzamba, Ronald Grant, David D. Eisenstat, Beverly Wilson, Anat Erdreich-Epstein, Almos Klekner, A. Sorana Morrissy, Richard Grundy, Young Shin Ra, Joanna J. Phillips, Alexandre Montpetit, Takafumi Wataya, Alexander R. Judkins, Shayna Zelcer, Nicholas K. Foreman, Rishi Lulla, Aline Cristiane Planello, Marc Remke, Harriet Druker, Annie Huang, Torsten Pietsch, José Pimentel, Jordan R. Hansford, Lindsey M. Hoffman, Mark Barszczyk, Tarik Tihan, Eugene Hwang, Vivek Mehta, László Bognár, Louis Letourneau, Donna L. Johnston, Stephen Yip, Lucie Lafay-Cousin, Mei Lu, Pasqualino De Antonellis, Katrin Scheinemann, Deena M.A. Gendoo, Shengrui Feng, James T. Rutka, G. Yancey Gillespie, Ho Keung Ng, Robert Hammond, David Malkin, Lúcia Roque, Anne Sophie Carret, King Ching Ho, Helen Toledano, Jennifer A. Chan, Monika Warmuth-Metz, Jacek Majewski, Jonathon Torchia, Livia Garzia, Stefan Rutkowski, Gino R. Somers, Tibor Hortobágyi, Ute Bartels, Peter Hauser, Ulrich Schüller, Cynthia Hawkins, Shih Hwa Chiou, Eric Bouffet, Adam Fleming, Alexandra N. Riemenschneider, Timothy E. Van Meter, Vijay Ramaswamy, Hideo Nakamura, Tiago Medina, Alexandre Vasiljevic, Noppadol Larbcharoensub, Patrick Sin-Chan, Christopher Dunham, Theodore Nicolaides, Iris Fried, Daniel Picard, Maryam Fouladi, Chris Fryer, Brian Golbourn, Mathieu Bourgey, Jean Michaud, Claudia C. Faria, Gary D. Bader, Mathieu Lupien, Amar Gajjar, Guillaume Bourque, Peter B. Dirks, Steffen Albrecht, Suradej Hongeng, Cheryl H. Arrowsmith, Uri Tabori, David A. Ramsay, Dalia Barsyte-Lovejoy, Paul Guilhamon, Michael Brudno, Nada Jabado, Juliette Hukin, Dong Anh Khuong-Quang, Michael D. Taylor, Tiffany Chan, Natalia R. Agamez, Daniel D. De Carvalho, Nongnuch Sirachainan, Samina Afzal, Seung Ah Choi, Diane K. Birks, Daniel W. Fults, Andrew S. Moore, Alyssa Reddy, Jason Fangusaro, Daniel Catchpoole, Yin Wang, Torchia, J., Golbourn, B., Feng, S., Ho, K. C., Sin-Chan, P., Vasiljevic, A., Norman, J. D., Guilhamon, P., Garzia, L., Agamez, N. R., Lu, M., Chan, T. S., Picard, D., de Antonellis, P., Khuong-Quang, D. -A., Planello, A. C., Zeller, C., Barsyte-Lovejoy, D., Lafay-Cousin, L., Letourneau, L., Bourgey, M., Yu, M., Gendoo, D. M. A., Dzamba, M., Barszczyk, M., Medina, T., Riemenschneider, A. N., Morrissy, A. S., Ra, Y. -S., Ramaswamy, V., Remke, M., Dunham, C. P., Yip, S., Ng, H. -K., Lu, J. -Q., Mehta, V., Albrecht, S., Pimentel, J., Chan, J. A., Somers, G. R., Faria, C. C., Roque, L., Fouladi, M., Hoffman, L. M., Moore, A. S., Wang, Y., Choi, S. A., Hansford, J. R., Catchpoole, D., Birks, D. K., Foreman, N. K., Strother, D., Klekner, A., Bognar, L., Garami, M., Hauser, P., Hortobagyi, T., Wilson, B., Hukin, J., Carret, A. -S., Van Meter, T. E., Hwang, E. I., Gajjar, A., Chiou, S. -H., Nakamura, H., Toledano, H., Fried, I., Fults, D., Wataya, T., Fryer, C., Eisenstat, D. D., Scheinemann, K., Fleming, A. J., Johnston, D. L., Michaud, J., Zelcer, S., Hammond, R., Afzal, S., Ramsay, D. A., Sirachainan, N., Hongeng, S., Larbcharoensub, N., Grundy, R. G., Lulla, R. R., Fangusaro, J. R., Druker, H., Bartels, U., Grant, R., Malkin, D., Mcglade, C. J., Nicolaides, T., Tihan, T., Phillips, J., Majewski, J., Montpetit, A., Bourque, G., Bader, G. D., Reddy, A. T., Gillespie, G. Y., Warmuth-Metz, M., Rutkowski, S., Tabori, U., Lupien, M., Brudno, M., Schuller, U., Pietsch, T., Judkins, A. R., Hawkins, C. E., Bouffet, E., Kim, S. -K., Dirks, P. B., Taylor, M. D., Erdreich-Epstein, A., Arrowsmith, C. H., De Carvalho, D. D., Rutka, J. T., Jabado, N., and Huang, A.

Subjects
Epigenomics, 0301 basic medicine, Cancer Research, Dasatinib, 1109 Neurosciences, 1112 Oncology and Carcinogenesis, ATRT, Epigenesis, Genetic, Central Nervous System Neoplasms, genomic, SMARCB1, Epigenesis, Central Nervous System Neoplasm, Teratoma, SMARCB1 Protein, Orvostudományok, Chromatin, 3. Good health, Oncology, DNA methylation, subgroup-specific therapeutic, Human, medicine.drug, Epigenomic, Cell Survival, Protein Kinase Inhibitor, Biology, Klinikai orvostudományok, Article, Receptor, Platelet-Derived Growth Factor beta, 03 medical and health sciences, Cell Line, Tumor, medicine, Humans, Oncology & Carcinogenesis, Epigenetics, Protein Kinase Inhibitors, rhabdoid tumor, Rhabdoid Tumor, Cell Proliferation, Cancer, DNA Methylation, medicine.disease, Pyrimidines, 030104 developmental biology, Pyrimidine, Mutation, Cancer research, enhancer
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

39. Perceptions of physicians caring for pediatric patients with cancer in Europe: insights into the use of palliative care, its timing, and barriers to early integration.

Author

Lacerda A, Bravo-Carretero IM, Ehrlich BS, Job G, Avilés Martínez M, Leiss U, Kokkinou G, Scheinemann K, Craig F, Krottendorfer K, Devidas M, Baker JN, Agulnik A, and McNeil MJ

Abstract

Background: Integrating pediatric palliative care (PPC) into pediatric oncology standard care is essential. Therefore, it is important to assess physicians' knowledge and perceptions of PPC to optimize its practice., Objective: To evaluate the knowledge, comfort levels, and perspectives of physicians regarding the timing and perceived barriers to integrating PPC into pediatric cancer care across Europe., Design: The Assessing Doctors' Attitudes on Palliative Treatment (ADAPT) survey, originally developed for other global regions, was culturally and contextually adapted for Europe., Setting/subjects: The survey was distributed via the European Society of Paediatric Oncology (SIOPE) membership listserv. Any physicians caring for children with cancer across Eastern, Southern, Central, and Northern Europe were invited to complete the survey., Results: A total of 198 physicians from 29 European countries completed the ADAPT survey. Physicians demonstrated relative agreement with the World Health Organization's guidance; median alignment was 83.4% (range 59.9%-94.1%). Although most respondents felt comfortable addressing physical (84.4%) and emotional (63.4%) needs, they felt less comfortable addressing spiritual needs (41.9%) and providing grief and bereavement support (48.5%). There were significant regional differences, such as physicians in Eastern and Southern Europe reporting a lack of PPC specialists, opioids, and home-based care, while those in Northern and Central Europe did not., Conclusion: Physicians caring for children with cancer throughout Europe have a good understanding of PPC. However, misconceptions about PPC persist, requiring educational and capacity-building efforts. Additionally, the regional differences in perceived barriers must be addressed to ensure equitable access to PPC for all European children with cancer., Competing Interests: The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest. The author(s) declared that they were an editorial board member of Frontiers, at the time of submission. This had no impact on the peer review process and the final decision., (Copyright © 2024 Lacerda, Bravo-Carretero, Ehrlich, Job, Avilés Martínez, Leiss, Kokkinou, Scheinemann, Craig, Krottendorfer, Devidas, Baker, Agulnik and McNeil.)

Published
2024
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40. Longitudinal needs and cancer knowledge in Swiss childhood cancer survivors transitioning from pediatric to adult follow-up care: results from the ACCS project.

Author

Buehlmann L, Otth M, and Scheinemann K

Abstract

Purpose: Childhood Cancer Survivors (CCSs) have an increased risk for treatment-related chronic health conditions, but the adherence to long-term follow-up (LTFU) care decreases over time. We therefore assessed the CCSs' development of cancer knowledge, cancer worries, self-management skills, and expectations for LTFU care in a structured, cancer center-based transition model-a crucial part for maintaining adherence., Methods: Using questionnaire-based surveys, we compared the CCSs' cancer knowledge with medical record data and assessed cancer worries (6 questions), self-management skills (15 questions), and expectations (12 questions) longitudinally by validated scales. We used descriptive statistics for presenting our results., Results: We analyzed 17 CCSs, 71% were female, had a median age of 8 years at diagnosis and 21 years at study enrollment. The knowledge about late effects increased during the transition process, except for the risk of secondary malignancies. Leukemia survivors had a decrease in cancer worries. At least 75% of the CCSs agreed to 11 of 15 self-management questions before and after transition, with the highest increase over time in less parental involvement. The CCSs expected the most, that physicians know the CCSs' cancer history, that the visit starts on time, and that physicians can always be called in case of questions., Conclusions: Our transition model improved cancer knowledge, especially the risk for late effects, decreased cancer worries, and identified expectations for LTFU care which should be considered in the future. A structured transition process with evidence-based tools further increases the knowledge of CCS for LTFU through empowerment., (© 2024. The Author(s).)

Published
2024
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41. The information needs of relatives of childhood cancer patients and survivors: A systematic review of quantitative evidence.

Author

Sievers Y, Roser K, Scheinemann K, Michel G, and Ilic A

Subjects
Humans, Child, Health Services Needs and Demand, Social Support, Parents psychology, Neoplasms, Cancer Survivors psychology, Family psychology, Needs Assessment
Abstract

Objective: We aimed to: (1) summarize the quantitative evidence on the information needs of relatives of childhood cancer patients, survivors, and children deceased from cancer; and (2) identify factors associated with these needs., Methods: PubMed, PsycINFO, Scopus, and CINAHL were systematically searched. The methodological quality of all included publications was assessed, and the extracted data were analyzed using narrative synthesis., Results: Of 5810 identified articles, 45 were included. Information needs were classified as unmet, met (satisfied), and unspecified and categorized into five domains: medical information, cancer-related consequences, lifestyle, family, and support. Most unmet information needs concerned cancer-related consequences (e.g., late effects), while information needs on support were generally met. Migrant background and higher education were associated with higher information needs among parents. Siblings had lower information needs than parents., Conclusion: This systematic review provides a comprehensive overview of the information needs of relatives in the context of childhood cancer, showing that information on cancer-related consequences is needed most often. The socioeconomic background of the relatives needs continued consideration throughout the cancer trajectory., Practice Implications: Our findings suggest the need for personalized information. Healthcare professionals should adapt their communication strategies to respond to the different and evolving needs of all affected relatives., Competing Interests: Declaration of Competing Interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2024 The Authors. Published by Elsevier B.V. All rights reserved.)

Published
2024
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42. Support Experiences and Wishes of Bereaved Parents After the Loss of Their Child to Cancer.

Author

Vokinger AK, Pedraza EC, Tinner EM, von Bueren AO, Scheinemann K, Bergstraesser E, Michel G, and De Clercq E

Subjects
Humans, Male, Female, Adult, Child, Middle Aged, Social Support, COVID-19 psychology, Child, Preschool, Qualitative Research, Adolescent, SARS-CoV-2, Infant, Parents psychology, Neoplasms psychology, Neoplasms therapy, Bereavement
Abstract

Introduction: The death of a child has a tremendous impact on parents' lives. The experience of parents who have lost a child to cancer may differ from other bereavement experiences, including other childhood and adulthood causes of death, because of the uncertainty of the prognosis, the aggressive treatment, and the potential for regret about treatment decisions. Bereavement care remains scarce, and effective interventions to meet the diverse needs of parents have not been defined., Objective: To provide insights on bereaved parents' experiences, their needs, and wishes of support following the loss of their child to cancer., Methods: We conducted 18 qualitative, in-depth, semi-structured interviews with 23 bereaved parents (seven fathers, 16 mothers), and used reflexive thematic analysis to analyze the data., Results: Parents received both informal and professional support. Regarding informal support, parents expressed a high level of ambivalence rooted in grief illiteracy. Parents also recognized their own struggles to express what forms of informal support they would have liked to receive. Support provided by healthcare professionals, institutions, and organizations involved in the children's care or in bereavement was inconsistent due to personnel time constraints, interpersonal relationships, or disruptions due to the COVID-19 pandemic. These factors could result in parents not having access to certain forms of support or not receiving long-term support., Conclusion: Improving grief literacy may strengthen informal support and make discussions of grief and death less taboo. Institutional policies, training, and networking may help to ensure that support provided by healthcare professionals, institutions, and organizations is less vulnerable to inconsistencies., (© 2024 The Author(s). Pediatric Blood & Cancer published by Wiley Periodicals LLC.)

Published
2025
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43. Quality criteria and certification for paediatric oncology centres: an international cross-sectional survey.

Author

Schladerer SP, Otth M, and Scheinemann K

Subjects
Humans, Cross-Sectional Studies, Surveys and Questionnaires, Cancer Care Facilities standards, Child, Quality of Health Care standards, Certification standards, Medical Oncology standards, Pediatrics standards
Abstract

Quality criteria and certification possibilities for paediatric oncology centres vary between countries and are not widely used. An overview of the type and how quality criteria and certifications are used in countries with highly developed healthcare systems is missing. This international cross-sectional survey investigated the use of quality criteria for paediatric oncology centres and whether certification is possible. We sent an online survey to paediatric oncologists from 32 countries worldwide and analysed the survey results and provided regional or national documents on quality criteria and certification possibilities descriptively. Paediatric oncologists from 28 (88%) countries replied. In most countries, the paediatric oncology centres were partly or completely grown historically (75%), followed by the development based on predefined criteria (29%), and due to political reason (25%), with more than one reason in some countries. Quality criteria are available in 20 countries (71%). We newly identified or specified five quality criteria, in addition to those from a previously performed systematic review. Certification of paediatric oncology centres is possible in 13 countries (46%), with a specific certification for paediatric oncology in seven, and a mandatory certification in three of them. The use of quality criteria and certification possibilities are heterogeneous, with quality criteria being more frequently used than certifications. Our study provides an overview of country-specific documents and links with quality criteria, and centre certification possibilities. It can serve as a reference document for stakeholders and may inform an international harmonization of quality criteria and centre certification between countries with similar healthcare systems., (© The Author(s) 2024. Published by Oxford University Press on behalf of International Society for Quality in Health Care.)

Published
2024
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44. Treatment of Medulloblastoma in the Adolescent and Young Adult Population: A Systematic Review.

Author

Otth M, Weiser A, Lee SY, Rudolf von Rohr L, Heesen P, Guerreiro Stucklin AS, and Scheinemann K

Abstract

Medulloblastoma is the most frequent high-grade tumor of the central nervous system in children but accounts for less than 1% of these tumors in adults. Adolescent and young adult (AYA) patients are between both age groups, and different approaches are used to treat medulloblastoma in this population. We performed a systematic review of studies published between 2007 and 2023 that reported treatment approaches and survival data of AYA patients with medulloblastoma, defined as 15 to 39 years of age at diagnosis. Due to the heterogeneity of data, a meta-analysis was not possible. Except for the omission of chemotherapy after radiotherapy in a few adult studies, the treatment backbone is very similar between studies starting enrolment during childhood and older adolescence or adulthood. Despite indications for a higher rate of early treatment termination due to toxicity in adults, survival data remain comparable between studies starting enrolment earlier or later in life. However, molecular subtyping was missing in most studies, so the survival data must be interpreted cautiously. Nevertheless, pediatric-inspired strategies in the AYA population are feasible, but individual dose adjustments may be necessary during treatment and should be considered upfront. Collaborative studies investigating the best treatment approach for medulloblastoma in the AYA population are needed in the future.

Published
2024
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45. Aftercare of Childhood Cancer Survivors in Switzerland-The General Practitioner Model.

Author

Otth M, Kroiss-Benninger S, and Scheinemann K

Abstract

Purpose: Childhood cancer survivors (CCS) represent a growing population worldwide, and lifelong follow-up care is recommended for most. Once CCS become adults, the transition to adult care is emerging. Today, there is no transition or long-term follow-up care model in the adult setting that clearly outweighs others. We therefore aimed to evaluate the transition to physicians outside the hospital. Methods: In this single-center, cross-sectional, questionnaire-based study, we assessed in 2022 the current follow-up care situation of CCS who already transitioned to physicians outside the hospital (family physicians, pediatricians). We asked CCS about cancer knowledge, worries, self-management skills, and expectations and physicians about their experience with CCS and their needs when caring for CCS. We included physicians where a CCS was transitioned to. We compared the results with CCS transitioned in a hospital setting and used descriptive statistics. Results: Twenty-three CCS responded to the questionnaire (median age at questionnaire of 22 years, median 14 years since diagnosis). Nearly two-thirds reported not being in follow-up care anymore. The cancer knowledge was good, and cancer worries were low. Twenty-eight physicians responded with 21 reporting that they care for CCS. Half of them see CCS for acute problems only. Physicians are open to care for CCS but request the necessary recommendations and would also be available for respective training. Conclusion: Transition to physicians might be an option for selected CCS. However, education and empowerment of CCS early on and education of physicians is urgently needed to prevent loss to follow-up, which may lead to lifelong nonengagement and incorrect perceptions about future health.

Published
2024
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46. Variant ALK-fusion positive anaplastic large cell lymphoma (ALCL): A population-based paediatric study of the NHL-BFM study group.

Author

Luedersen J, Stadt UZ, Richter J, Oschlies I, Klapper W, Rosenwald A, Kalinova M, Simonitsch-Klupp I, Siebert R, Zimmermann M, Qi M, Nakel J, Scheinemann K, Knörr F, Attarbaschi A, Kabickova E, Woessmann W, and Damm-Welk C

Subjects
Humans, Child, Male, Female, Adolescent, Child, Preschool, Oncogene Proteins, Fusion genetics, Prognosis, Nuclear Proteins genetics, Nuclear Proteins metabolism, Infant, Tropomyosin, Lymphoma, Large-Cell, Anaplastic genetics, Lymphoma, Large-Cell, Anaplastic pathology, Anaplastic Lymphoma Kinase genetics, Anaplastic Lymphoma Kinase analysis, Nucleophosmin
Abstract

Frequency, distribution and prognostic meaning of ALK-partner genes other than NPM1 in ALK-positive anaplastic large-cell lymphoma (ALCL) are unknown. Forty-nine of 316 ALCL diagnosed in the NHL-BFM study group showed no nuclear ALK expression suggestive of a variant ALK-partner; 41 were analysed by genomic capture high-throughput sequencing or specific RT-PCRs. NPM1::ALK was detected in 13 cases. Among the 28 patients with a non-NPM1::ALK-fusion partner, ATIC (n = 8; 29%) and TPM3 (n = 9; 32%) were the most common. Five of eight patients with ATIC::ALK-positive ALCL relapsed, none of nine with TPM3::ALK. Variant ALK-partners are rare and potentially associated with different prognoses., (© 2024 The Authors. British Journal of Haematology published by British Society for Haematology and John Wiley & Sons Ltd.)

Published
2024
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47. Late Adverse Effects after Treatment for Childhood Acute Leukemia.

Author

Roganovic J, Haupt R, Bárdi E, Hjorth L, Michel G, Pavasovic V, Scheinemann K, van der Pal HJ, Zadravec Zaletel L, Amariutei AE, and Skinner R

Subjects
Humans, Child, Hematopoietic Stem Cell Transplantation adverse effects, Long Term Adverse Effects chemically induced, Antineoplastic Agents adverse effects, Leukemia, Myeloid, Acute therapy, Leukemia, Myeloid, Acute drug therapy, Precursor Cell Lymphoblastic Leukemia-Lymphoma drug therapy, Precursor Cell Lymphoblastic Leukemia-Lymphoma therapy, Cancer Survivors
Abstract

The aim of this review is to raise awareness and knowledge among healthcare professionals and policymakers about late adverse effects in survivors of childhood leukemia. With contemporary treatment, over 90% of children with acute lymphoblastic leukemia (ALL) and over 60% with acute myeloid leukemia (AML) are cured. Large cohort studies demonstrate that 20% of ALL and most AML survivors have at least one chronic health condition by 20-25 years after diagnosis. These are life-changing or threatening in some survivors and contribute to increased premature mortality. We describe the frequency, causes, clinical features, and natural history of the most frequent and severe late adverse effects in childhood leukemia survivors, including subsequent malignant neoplasms, metabolic toxicity, gonadotoxicity and impaired fertility, endocrinopathy and growth disturbances, bone toxicity, central and peripheral neurotoxicity, cardiotoxicity, psychosocial late effects, accelerated ageing and late mortality. The wide range of late effects in survivors of haemopoietic stem cell transplant is highlighted. Recent developments informing the approach to long-term survivorship care are discussed, including electronic personalized patient-specific treatment summaries and care plans such as the Survivor Passport (SurPass), surveillance guidelines and models of care. The importance of ongoing vigilance is stressed given the increasing use of novel targeted drugs with limited experience of long-term outcomes. CONCLUSION: It is vital to raise awareness of the existence and severity of late effects of childhood leukemia therapy among parents, patients, health professionals, and policymakers. Structured long-term surveillance recommendations are necessary to standardize follow-up care., (Copyright © 2024 Roganovic et al. This article is available under a Creative Commons License (Attribution 4.0 International).)

Published
2024
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48. Incidence and prevalence of musculoskeletal health conditions in survivors of childhood and adolescent cancers: A report from the Swiss childhood cancer survivor study.

Author

Christen S, Roser K, Mader L, Otth M, Scheinemann K, Sommer G, Kuehni C, and Michel G

Subjects
Humans, Adolescent, Female, Male, Young Adult, Incidence, Switzerland epidemiology, Prevalence, Adult, Middle Aged, Child, Registries, Cancer Survivors statistics & numerical data, Musculoskeletal Diseases epidemiology, Musculoskeletal Diseases etiology, Neoplasms epidemiology
Abstract

Purpose: Childhood cancer and its treatment can cause damage to the musculoskeletal system. We aimed to determine the incidence and prevalence of musculoskeletal health conditions (MSHC) in survivors, and to investigate differences by cancer-related characteristics., Methods: We used data from the Childhood Cancer Registry and the Swiss Childhood Cancer Survivor Study, including survivors (≥5 years since diagnosis; diagnosed 1976-2015 at <20 years of age) aged ≥15 years at study. Cumulative incidence and prevalence of MSHCs (osteoporosis, limb length discrepancy, limited joint mobility, bone/joint pain, scoliosis, changes to chest/ribs and amputation) were calculated from self-reported data., Results: We included 2645 survivors (53% men; median age 24 years, range 15-59 years). Prevalence and cumulative incidence of any MSHC was 21% and 26%, respectively. Incidence rate for any MSHC was 15.6/1000 person-years. Scoliosis (8%), bone/joint pain (7%) and limited joint mobility (7%) were the most prevalent MSHC. MSHC co-occurred with other health conditions in 87% of survivors. We found increased rates of MSHC in women (RR = 1.4, 95%CI: 1.2-1.7), bone tumour survivors (RR = 6.0, 95%CI: 4.5-7.9), survivors older at diagnosis (11-15 years: RR = 1.8, 95%CI: 1.5-2.3), after a relapse (RR = 1.5, 95%CI: 1.3-1.9), treatment with surgery (RR = 1.2, 95%CI: 1.0-1.5), chemotherapy (RR = 1.4, 95%CI: 1.1-1.8) or stem cell transplantation (RR = 1.6, 95%CI: 1.0-2.5), and more recent year of diagnosis (2011-2015: RR = 4.3, 95%CI: 2.8-6.8)., Conclusion: MSHCs are prevalent in survivors, the risk is increasing in younger survivor cohorts, and MSHCs usually occur in multimorbid survivors. Strengthening of rehabilitation services and appropriate referrals are needed to mitigate the effects of the cancer and cancer treatment., (© 2024 The Authors. Cancer Medicine published by John Wiley & Sons Ltd.)

Published
2024
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49. Exploring grandparents' psychosocial responses to childhood cancer: A qualitative study.

Author

Holmer P, Muehlebach N, Ilic A, Priboi C, Roser K, Raguindin PF, Tinner EM, Baechtold R, Ansari M, Diezi M, Lemmel E, Schilling F, Farrag A, Scheinemann K, and Michel G

Subjects
Child, Humans, Female, Aged, Male, Family psychology, Anxiety, Coping Skills, Grandparents psychology, Neoplasms psychology
Abstract

Objective: A childhood cancer diagnosis is a traumatic experience for patients and their families. However, little is known about the effect on grandparents. We aimed to investigate the negative psychosocial impact, coping strategies, and positive outcomes of grandparents of childhood cancer patients in Switzerland., Methods: We collected data using a semi-structured interview guide and applied qualitative content analysis., Results: We conducted 20 interviews with 23 grandparents (57% female; mean age = 66.9 years; SD = 6.4; range = 57.0-82.4) of 13 affected children (69% female; mean age = 7.5 years; SD = 6.1; range = 1.0-18.9) between January 2022 and April 2023. The mean time since diagnosis was 1.0 years (SD = 0.5; range = 0.4-1.9). Grandparents were in shock and experienced strong feelings of fear and helplessness. They were particularly afraid of a relapse or late effects. The worst part for most was seeing their grandchild suffer. Many stated that their fear was always present which could lead to tension and sleep problems. To cope with these negative experiences, the grandparents used internal and external strategies, such as accepting the illness or talking to their spouse and friends. Some grandparents also reported positive outcomes, such as getting emotionally closer to family members and appreciating things that had previously been taken for granted., Conclusions: Grandparents suffer greatly when their grandchild is diagnosed with cancer. Encouragingly, most grandparents also reported coping strategies and positive outcomes despite the challenges. Promoting coping strategies and providing appropriate resources could reduce the psychological burden of grandparents and strengthen the whole family system., (© 2024 The Authors. Psycho-Oncology published by John Wiley & Sons Ltd.)

Published
2024
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50. LOGGIC/FIREFLY-2: a phase 3, randomized trial of tovorafenib vs. chemotherapy in pediatric and young adult patients with newly diagnosed low-grade glioma harboring an activating RAF alteration.

Author

van Tilburg CM, Kilburn LB, Perreault S, Schmidt R, Azizi AA, Cruz-Martínez O, Zápotocký M, Scheinemann K, Meeteren AYNS, Sehested A, Opocher E, Driever PH, Avula S, Ziegler DS, Capper D, Koch A, Sahm F, Qiu J, Tsao LP, Blackman SC, Manley P, Milde T, Witt R, Jones DTW, Hargrave D, and Witt O

Subjects
Animals, Child, Humans, Young Adult, Proto-Oncogene Proteins B-raf, Treatment Outcome, Mutation, Mitogen-Activated Protein Kinases, Oximes, Pyridones, Pyrimidinones therapeutic use, Antineoplastic Combined Chemotherapy Protocols therapeutic use, Fireflies metabolism, Glioma drug therapy, Glioma genetics, Glioma metabolism
Abstract

Background: Pediatric low-grade glioma (pLGG) is essentially a single pathway disease, with most tumors driven by genomic alterations affecting the mitogen-activated protein kinase/ERK (MAPK) pathway, predominantly KIAA1549::BRAF fusions and BRAF V600E mutations. This makes pLGG an ideal candidate for MAPK pathway-targeted treatments. The type I BRAF inhibitor, dabrafenib, in combination with the MEK inhibitor, trametinib, has been approved by the United States Food and Drug Administration for the systemic treatment of BRAF V600E-mutated pLGG. However, this combination is not approved for the treatment of patients with tumors harboring BRAF fusions as type I RAF inhibitors are ineffective in this setting and may paradoxically enhance tumor growth. The type II RAF inhibitor, tovorafenib (formerly DAY101, TAK-580, MLN2480), has shown promising activity and good tolerability in patients with BRAF-altered pLGG in the phase 2 FIREFLY-1 study, with an objective response rate (ORR) per Response Assessment in Neuro-Oncology high-grade glioma (RANO-HGG) criteria of 67%. Tumor response was independent of histologic subtype, BRAF alteration type (fusion vs. mutation), number of prior lines of therapy, and prior MAPK-pathway inhibitor use., Methods: LOGGIC/FIREFLY-2 is a two-arm, randomized, open-label, multicenter, global, phase 3 trial to evaluate the efficacy, safety, and tolerability of tovorafenib monotherapy vs. current standard of care (SoC) chemotherapy in patients < 25 years of age with pLGG harboring an activating RAF alteration who require first-line systemic therapy. Patients are randomized 1:1 to either tovorafenib, administered once weekly at 420 mg/m 2 (not to exceed 600 mg), or investigator's choice of prespecified SoC chemotherapy regimens. The primary objective is to compare ORR between the two treatment arms, as assessed by independent review per RANO-LGG criteria. Secondary objectives include comparisons of progression-free survival, duration of response, safety, neurologic function, and clinical benefit rate., Discussion: The promising tovorafenib activity data, CNS-penetration properties, strong scientific rationale combined with the manageable tolerability and safety profile seen in patients with pLGG led to the SIOPe-BTG-LGG working group to nominate tovorafenib for comparison with SoC chemotherapy in this first-line phase 3 trial. The efficacy, safety, and functional response data generated from the trial may define a new SoC treatment for newly diagnosed pLGG., Trial Registration: ClinicalTrials.gov: NCT05566795. Registered on October 4, 2022., (© 2024. The Author(s).)

Published
2024
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