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2. Outcomes in lung-only metastatic rhabdomyosarcoma: An analysis of data from the European paediatric Soft tissue sarcoma Study Group MTS 2008 study

Author

Chisholm, Julia C., Schoot, Reineke A., Cameron, Alison L., Casanova, Michela, Minard-Colin, Veronique, Coppadoro, Beatrice, Garrido, Marta, Rogers, Timothy, Orbach, Daniel, Glosli, Heidi, Ben-Arush, Miriam, Ferman, Sima, Scarzello, Giovanni, van Rijn, Rick R., Hladun, Raquel, Corradini, Nadege, Ferrari, Andrea, Jenney, Meriel, Bisogno, Gianni, and Merks, Johannes H.M.

Published
2023
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3. The Impact of Radiation Therapy in Children and Adolescents With Metastatic Rhabdomyosarcoma

Author

Cameron, Alison L., Elze, Markus C., Casanova, Michela, Geoerger, Birgit, Gaze, Mark N., Minard-Colin, Veronique, McHugh, Kieran, van Rijn, Rick R., Kelsey, Anna, Martelli, Hélène, Mandeville, Henry, Bisogno, Gianni, Lowis, Stephen, Ronghe, Milind, Orbach, Daniel, Guizani, Cecile, Fürst-Recktenwald, Sabine, Chisholm, Julia C., and Merks, Johannes H.M.

Published
2021
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6. Phase I results of a phase I/II study of weekly nab-paclitaxel in paediatric patients with recurrent/refractory solid tumours: A collaboration with innovative therapies for children with cancer

Author

Moreno, Lucas, Casanova, Michela, Chisholm, Julia C., Berlanga, Pablo, Chastagner, Pascal B., Baruchel, Sylvain, Amoroso, Loredana, Gallego Melcón, Soledad, Gerber, Nicolas U., Bisogno, Gianni, Fagioli, Franca, Geoerger, Birgit, Glade Bender, Julia L., Aerts, Isabelle, Bergeron, Christophe, Hingorani, Pooja, Elias, Ileana, Simcock, Mathew, Ferrara, Stefano, Le Bruchec, Yvan, Slepetis, Ruta, Chen, Nianhang, and Vassal, Gilles

Published
2018
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8. Open-label, multicentre, randomised, phase II study of the EpSSG and the ITCC evaluating the addition of bevacizumab to chemotherapy in childhood and adolescent patients with metastatic soft tissue sarcoma (the BERNIE study)

Author

Chisholm, Julia C., Merks, Johannes H.M., Casanova, Michela, Bisogno, Gianni, Orbach, Daniel, Gentet, Jean-Claude, Thomassin-Defachelles, Anne-Sophie, Chastagner, Pascal, Lowis, Stephen, Ronghe, Milind, McHugh, Kieran, van Rijn, Rick R., Hilton, Magalie, Bachir, Jeanette, Fürst-Recktenwald, Sabine, Geoerger, Birgit, and Oberlin, Odile

Published
2017
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10. Intra‐abdominal desmoplastic small round cell tumor: The European pediatric Soft tissue sarcoma Study Group (E p SSG) experience

Author

Berlanga, Pablo, primary, Orbach, Daniel, additional, Schoot, Reineke A., additional, Casanova, Michela, additional, Alaggio, Rita, additional, Corradini, Nadege, additional, Brennan, Bernadette, additional, Ramirez‐Villar, Gema L., additional, Hjalgrim, Lisa Lyngsie, additional, Chisholm, Julia C., additional, Bisogno, Gianni, additional, Coppadoro, Beatrice, additional, Safwat, Akmal, additional, Merks, Johannes H. M., additional, Burrieza, Gabriela Guillen, additional, van Noesel, Max M., additional, and Ferrari, Andrea, additional

Published
2023
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11. Metastatic adult‐type non‐rhabdomyosarcoma soft tissue sarcomas in children and adolescents: A cohort study from the European paediatric Soft tissue sarcoma Study Group

Author

Ferrari, Andrea, primary, Orbach, Daniel, additional, Casanova, Michela, additional, van Noesel, Max M., additional, Berlanga, Pablo, additional, Brennan, Bernadette, additional, Corradini, Nadege, additional, Schoot, Reineke A., additional, Ramirez‐Villar, Gema L., additional, Hjalgrim, Lisa Lyngsie, additional, Alaggio, Rita, additional, Guillen Burrieza, Gabriela, additional, Safwat, Akmal, additional, Cameron, Alison L., additional, van Rijn, Rick R., additional, Minard‐Colin, Veronique, additional, Zanetti, Ilaria, additional, Bisogno, Gianni, additional, Chisholm, Julia C., additional, and Merks, Johannes H. M., additional

Published
2023
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12. Outcomes in lung-only metastatic rhabdomyosarcoma: An analysis of data from the European paediatric Soft tissue sarcoma Study Group MTS 2008 study

Author

Arts-assistenten Kinderen, Onderzoek Beeld, Chisholm, Julia C., Schoot, Reineke A., Cameron, Alison L., Casanova, Michela, Minard-Colin, Veronique, Coppadoro, Beatrice, Garrido, Marta, Rogers, Timothy, Orbach, Daniel, Glosli, Heidi, Ben-Arush, Miriam, Ferman, Sima, Scarzello, Giovanni, van Rijn, Rick R., Hladun, Raquel, Corradini, Nadege, Ferrari, Andrea, Jenney, Meriel, Bisogno, Gianni, Merks, Johannes H.M., Arts-assistenten Kinderen, Onderzoek Beeld, Chisholm, Julia C., Schoot, Reineke A., Cameron, Alison L., Casanova, Michela, Minard-Colin, Veronique, Coppadoro, Beatrice, Garrido, Marta, Rogers, Timothy, Orbach, Daniel, Glosli, Heidi, Ben-Arush, Miriam, Ferman, Sima, Scarzello, Giovanni, van Rijn, Rick R., Hladun, Raquel, Corradini, Nadege, Ferrari, Andrea, Jenney, Meriel, Bisogno, Gianni, and Merks, Johannes H.M.

Published
2023

13. Intra-abdominal desmoplastic small round cell tumor: The European pediatric Soft tissue sarcoma Study Group (EpSSG) experience

Author

Onderzoek Beeld, Cancer, Berlanga, Pablo, Orbach, Daniel, Schoot, Reineke A, Casanova, Michela, Alaggio, Rita, Corradini, Nadege, Brennan, Bernadette, Ramirez-Villar, Gema L, Hjalgrim, Lisa Lyngsie, Chisholm, Julia C, Bisogno, Gianni, Coppadoro, Beatrice, Safwat, Akmal, Merks, Johannes H M, Burrieza, Gabriela Guillen, van Noesel, Max M, Ferrari, Andrea, Onderzoek Beeld, Cancer, Berlanga, Pablo, Orbach, Daniel, Schoot, Reineke A, Casanova, Michela, Alaggio, Rita, Corradini, Nadege, Brennan, Bernadette, Ramirez-Villar, Gema L, Hjalgrim, Lisa Lyngsie, Chisholm, Julia C, Bisogno, Gianni, Coppadoro, Beatrice, Safwat, Akmal, Merks, Johannes H M, Burrieza, Gabriela Guillen, van Noesel, Max M, and Ferrari, Andrea

Published
2023

14. Metastatic adult-type non-rhabdomyosarcoma soft tissue sarcomas in children and adolescents: A cohort study from the European paediatric Soft tissue sarcoma Study Group

Author

Onderzoek Beeld, Cancer, PMC Medisch specialisten, Ferrari, Andrea, Orbach, Daniel, Casanova, Michela, van Noesel, Max M, Berlanga, Pablo, Brennan, Bernadette, Corradini, Nadege, Schoot, Reineke A, Ramirez-Villar, Gema L, Hjalgrim, Lisa Lyngsie, Alaggio, Rita, Guillen Burrieza, Gabriela, Safwat, Akmal, Cameron, Alison L, van Rijn, Rick R, Minard-Colin, Veronique, Zanetti, Ilaria, Bisogno, Gianni, Chisholm, Julia C, Merks, Johannes H M, Onderzoek Beeld, Cancer, PMC Medisch specialisten, Ferrari, Andrea, Orbach, Daniel, Casanova, Michela, van Noesel, Max M, Berlanga, Pablo, Brennan, Bernadette, Corradini, Nadege, Schoot, Reineke A, Ramirez-Villar, Gema L, Hjalgrim, Lisa Lyngsie, Alaggio, Rita, Guillen Burrieza, Gabriela, Safwat, Akmal, Cameron, Alison L, van Rijn, Rick R, Minard-Colin, Veronique, Zanetti, Ilaria, Bisogno, Gianni, Chisholm, Julia C, and Merks, Johannes H M

Published
2023

15. Intra‐abdominal desmoplastic small round cell tumor: The European pediatric Soft tissue sarcoma Study Group (EpSSG) experience.

Author

Berlanga, Pablo, Orbach, Daniel, Schoot, Reineke A., Casanova, Michela, Alaggio, Rita, Corradini, Nadege, Brennan, Bernadette, Ramirez‐Villar, Gema L., Hjalgrim, Lisa Lyngsie, Chisholm, Julia C., Bisogno, Gianni, Coppadoro, Beatrice, Safwat, Akmal, Merks, Johannes H. M., Burrieza, Gabriela Guillen, van Noesel, Max M., and Ferrari, Andrea

Published
2023
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17. Metastatic rhabdomyosarcoma with exclusive distant lymph node involvement: A European Pediatric Soft tissue sarcoma Study Group (EpSSG) report

Author

Mercolini, Federico, primary, Merks, Johannes H. M., additional, Minard‐Colin, Veronique, additional, Cameron, Alison, additional, van Scheltinga, Scheila E. J. Terwisscha, additional, Sher, Osnat, additional, Fichera, Giulia, additional, Orbach, Daniel, additional, Glosli, Heidi, additional, Coppadoro, Beatrice, additional, Gallego, Soledad, additional, Chisholm, Julia C., additional, and Bisogno, Gianni, additional

Published
2022
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18. Shedding a Light on the Challenges of Adolescents and Young Adults with Rhabdomyosarcoma

Author

Ferrari, Andrea, primary, Gatz, Susanne Andrea, additional, Minard-Colin, Veronique, additional, Alaggio, Rita, additional, Hovsepyan, Shushan, additional, Orbach, Daniel, additional, Gasparini, Patrizia, additional, Defachelles, Anne-Sophie, additional, Casanova, Michela, additional, Milano, Giuseppe Maria, additional, Chisholm, Julia C., additional, Jenney, Meriel, additional, Bisogno, Gianni, additional, Rogers, Timothy, additional, Mandeville, Henry C., additional, Shipley, Janet, additional, Miah, Aisha B., additional, Merks, Johannes H. M., additional, and van der Graaf, Winette T. A., additional

Published
2022
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19. Metastatic Rhabdomyosarcoma: Results of the European Paediatric Soft Tissue Sarcoma Study Group MTS 2008 Study and Pooled Analysis With the Concurrent BERNIE Study

Author

Schoot, Reineke A., primary, Chisholm, Julia C., additional, Casanova, Michela, additional, Minard-Colin, Veronique, additional, Geoerger, Birgit, additional, Cameron, Alison L., additional, Coppadoro, Beatrice, additional, Zanetti, Ilaria, additional, Orbach, Daniel, additional, Kelsey, Anna, additional, Rogers, Timothy, additional, Guizani, Cecile, additional, Elze, Markus, additional, Ben-Arush, Myriam, additional, McHugh, Kieran, additional, van Rijn, Rick R., additional, Ferman, Sima, additional, Gallego, Soledad, additional, Ferrari, Andrea, additional, Jenney, Meriel, additional, Bisogno, Gianni, additional, and Merks, Johannes H.M., additional

Published
2022
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20. Molecular Characterization of Circulating Tumor DNA in Pediatric Rhabdomyosarcoma: A Feasibility Study

Author

Ruhen, Olivia, primary, Lak, Nathalie S.M., additional, Stutterheim, Janine, additional, Danielli, Sara G., additional, Chicard, Mathieu, additional, Iddir, Yasmine, additional, Saint-Charles, Alexandra, additional, Di Paolo, Virginia, additional, Tombolan, Lucia, additional, Gatz, Susanne A., additional, Aladowicz, Ewa, additional, Proszek, Paula, additional, Jamal, Sabri, additional, Stankunaite, Reda, additional, Hughes, Deborah, additional, Carter, Paul, additional, Izquierdo, Elisa, additional, Wasti, Ajla, additional, Chisholm, Julia C., additional, George, Sally L., additional, Pace, Erika, additional, Chesler, Louis, additional, Aerts, Isabelle, additional, Pierron, Gaelle, additional, Zaidi, Sakina, additional, Delattre, Olivier, additional, Surdez, Didier, additional, Kelsey, Anna, additional, Hubank, Michael, additional, Bonvini, Paolo, additional, Bisogno, Gianni, additional, Di Giannatale, Angela, additional, Schleiermacher, Gudrun, additional, Schäfer, Beat W., additional, Tytgat, Godelieve A.M., additional, and Shipley, Janet, additional

Published
2022
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22. Prognostic relevance of early radiologic response to induction chemotherapy in pediatric rhabdomyosarcoma: A report from the International Society of Pediatric Oncology Malignant Mesenchymal Tumor 95 study

Author

Vaarwerk, Bas, van der Lee, Johanna H., Breunis, Willemijn B., Orbach, Daniel, Chisholm, Julia C., Cozic, Nathalie, Jenney, Meriel, van Rijn, Rick R., McHugh, Kieran, Gallego, Soledad, Glosli, Heidi, Devalck, Christine, Gaze, Mark N., Kelsey, Anna, Bergeron, Christophe, Stevens, Michael C. G., Oberlin, Odile, Minard‐Colin, Veronique, and Merks, Johannes H. M.

Published
2018
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23. Adolescents and young adults with rhabdomyosarcoma treated in the European paediatric Soft tissue sarcoma Study Group (EpSSG) protocols: a cohort study

Author

Ferrari, Andrea, primary, Chisholm, Julia C, additional, Jenney, Meriel, additional, Minard-Colin, Veronique, additional, Orbach, Daniel, additional, Casanova, Michela, additional, Guillen, Gabriela, additional, Glosli, Heidi, additional, van Rijn, Rick R, additional, Schoot, Reineke A, additional, Cameron, Alison L, additional, Rogers, Timothy, additional, Alaggio, Rita, additional, Ben-Arush, Myriam, additional, Mandeville, Henry C, additional, Devalck, Christine, additional, Defachelles, Anne-Sophie, additional, Coppadoro, Beatrice, additional, Bisogno, Gianni, additional, and Merks, Johannes H M, additional

Published
2022
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24. Shedding a Light on the Challenges of Adolescents and Young Adults with Rhabdomyosarcoma

Author

Ferrari, Andrea, Gatz, Susanne Andrea, Minard-Colin, Veronique, Alaggio, Rita, Hovsepyan, Shushan, Orbach, Daniel, Gasparini, Patrizia, Defachelles, Anne Sophie, Casanova, Michela, Milano, Giuseppe Maria, Chisholm, Julia C., Jenney, Meriel, Bisogno, Gianni, Rogers, Timothy, Mandeville, Henry C., Shipley, Janet, Miah, Aisha B., Merks, Johannes H.M., van der Graaf, Winette T.A., Ferrari, Andrea, Gatz, Susanne Andrea, Minard-Colin, Veronique, Alaggio, Rita, Hovsepyan, Shushan, Orbach, Daniel, Gasparini, Patrizia, Defachelles, Anne Sophie, Casanova, Michela, Milano, Giuseppe Maria, Chisholm, Julia C., Jenney, Meriel, Bisogno, Gianni, Rogers, Timothy, Mandeville, Henry C., Shipley, Janet, Miah, Aisha B., Merks, Johannes H.M., and van der Graaf, Winette T.A.

Abstract

Rhabdomyosarcoma (RMS) is a typical tumour of childhood but can occur at any age. Several studies have reported that adolescent and young adult (AYA) patients with RMS have poorer survival than do younger patients. This review discusses the specific challenges in AYA patients with pediatric-type RMS, exploring possible underlying factors which may influence different outcomes. Reasons for AYA survival gap are likely multifactorial, and might be related to differences in tumor biology and intrinsic aggressiveness, or differences in clinical management (that could include patient referral patterns, time to diagnosis, enrolment into clinical trials, the adequacy and intensity of treatment), as well as patient factors (including physiology and comorbidity that may influence treatment tolerability, drug pharmacokinetics and efficacy). However, improved survival has been reported in the most recent studies for AYA patients treated on pediatric RMS protocols. Different strategies may help to further improve outcome, such as supporting trans-age academic societies and national/international collaborations; developing specific clinical trials without upper age limit; defining integrated and comprehensive approach to AYA patients, including the genomic aspects; establishing multidisciplinary tumor boards with involvement of both pediatric and adult oncologists to discuss all pediatric-type RMS patients; developing dedicated projects with specific treatment recommendations and registry/database.

Published
2022

26. Reply to H. B et al

Author

Defachelles, Anne-Sophie, primary, Bogart, Emilie, additional, Casanova, Michela, additional, Merks, Johannes H.M., additional, Bisogno, Gianni, additional, Calareso, Giuseppina, additional, Melcon, Soledad Gallego, additional, Gatz, Susanne Andrea, additional, Le Deley, Marie-Cécile, additional, McHugh, Kieran, additional, Probst, Alicia, additional, Rocourt, Nathalie, additional, van Rijn, Rick R., additional, Minard-Colin, Véronique, additional, and Chisholm, Julia C., additional

Published
2022
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27. Circulating tumour DNA sequencing to determine therapeutic response and identify tumour heterogeneity in patients with paediatric solid tumours

Author

Stankunaite, Reda, primary, George, Sally L., additional, Gallagher, Lewis, additional, Jamal, Sabri, additional, Shaikh, Ridwan, additional, Yuan, Lina, additional, Hughes, Debbie, additional, Proszek, Paula Z., additional, Carter, Paul, additional, Pietka, Grzegorz, additional, Heide, Timon, additional, James, Chela, additional, Tari, Haider, additional, Lynn, Claire, additional, Jain, Neha, additional, Portela, Laura Rey, additional, Rogers, Tony, additional, Vaidya, Sucheta J., additional, Chisholm, Julia C., additional, Carceller, Fernando, additional, Szychot, Elwira, additional, Mandeville, Henry, additional, Angelini, Paola, additional, Jesudason, Angela B., additional, Jackson, Michael, additional, Marshall, Lynley V., additional, Gatz, Susanne A., additional, Anderson, John, additional, Sottoriva, Andrea, additional, Chesler, Louis, additional, and Hubank, Michael, additional

Published
2022
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30. Transforming training into practice with the conflict management framework: a mixed methods study

Author

Lyons, Oscar, primary, Forbat, Liz, additional, Menson, Esse, additional, Chisholm, Julia C, additional, Pryde, Kate, additional, Conlin, Siobhan, additional, Felton, Victoria, additional, Ingle, Susanne, additional, McKenzie, Celia, additional, Ramachandran, Rohana, additional, Sayer, Charlotte, additional, Snowball, Carly, additional, Strachan-Gadsby, Emma, additional, Tisovszky, Natasha, additional, and Barclay, Sarah, additional

Published
2021
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31. Randomized Phase II Trial of Vincristine-Irinotecan With or Without Temozolomide, in Children and Adults With Relapsed or Refractory Rhabdomyosarcoma: A European Paediatric Soft Tissue Sarcoma Study Group and Innovative Therapies for Children With Cancer Trial

Author

Defachelles, Anne-Sophie, primary, Bogart, Emilie, additional, Casanova, Michela, additional, Merks, Johannes H. M., additional, Bisogno, Gianni, additional, Calareso, Giuseppina, additional, Gallego Melcon, Soledad, additional, Gatz, Susanne Andrea, additional, Le Deley, Marie-Cécile, additional, McHugh, Kieran, additional, Probst, Alicia, additional, Rocourt, Nathalie, additional, van Rijn, Rick R., additional, Wheatley, Keith, additional, Minard-Colin, Véronique, additional, and Chisholm, Julia C., additional

Published
2021
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32. Optimising Antimicrobial Selection and Duration in the Treatment of Febrile Neutropenia in Children

Author

Morgan,Jessica E, Phillips,Bob, Haeusler,Gabrielle M, and Chisholm,Julia C

Subjects
Infection and Drug Resistance
Abstract

Jessica E Morgan,1,2 Bob Phillips,1,2 Gabrielle M Haeusler,3– 6 Julia C Chisholm7 1Centre for Reviews and Dissemination, University of York, Heslington, YO10 5DD, UK; 2Department of Paediatric Oncology, Leeds Teaching Hospitals NHS Trust, Leeds, LS1 3EX, UK; 3NHMRC National Centre for Infections in Cancer, Sir Peter MacCallum Department of Oncology, University of Melbourne, Parkville, Victoria, 3010, Australia; 4Sir Peter MacCallum Department of Oncology, University of Melbourne, Parkville, Victoria, 3010, Australia; 5Infection Diseases Unit, Department of General Medicine, Royal Children’s Hospital, Parkville, Victoria, 3168, Australia; 6Murdoch Children’s Research Institute, Parkville, Victoria, 3052, Australia; 7Royal Marsden Hospital and Institute of Cancer Research, Sutton, SM2 5PT, UKCorrespondence: Jessica E MorganCentre for Reviews and Dissemination, University of York, Heslington, YO10 5DD, UKTel +44 01904 321082Email jess.morgan@york.ac.ukAbstract: Febrile neutropenia (FN) is a frequent complication of cancer treatment in children. Owing to the potential for overwhelming bacterial sepsis, the recognition and management of FN requires rapid implementation of evidenced-based management protocols. Treatment paradigms have progressed from hospitalisation with broad spectrum antibiotics for all patients, through to risk adapted approaches to management. Such risk adapted approaches aim to provide safe care through incorporating antimicrobial stewardship (AMS) principles such as implementation of comprehensive clinical pathways incorporating de-escalation strategies with the imperative to reduce hospital stay and antibiotic exposure where possible in order to improve patient experience, reduce costs and diminish the risk of nosocomial infection. This review summarises the principles of risk stratification in FN, the current key considerations for optimising empiric antimicrobial selection including knowledge of antimicrobial resistance patterns and emerging technologies for rapid diagnosis of specific infections and summarises existing evidence on time to treatment, investigations required and duration of treatment. To aid treating physicians we suggest the key features based on current evidence that should be part of any FN management guideline and highlight areas for future research. The focus is on treatment of bacterial infections although fungal and viral infections are also important in this patient group.Keywords: paediatric, febrile neutropenia, antimicrobials

Published
2021

33. Metastatic Rhabdomyosarcoma: Results of the European Soft Tissue Sarcoma Study Group MTS 2008 Study and Pooled Analysis With the Concurrent BERNIE Study.

Author

Schoot, Reineke A., Chisholm, Julia C., Casanova, Michela, Minard-Colin, Veronique, Geoerger, Birgit, Cameron, Alison L., Coppadoro, Beatrice, Zanetti, Ilaria, Orbach, Daniel, Kelsey, Anna, Rogers, Timothy, Guizani, Cecile, Elze, Markus, Ben-Arush, Myriam, McHugh, Kieran, van Rijn, Rick R., Ferman, Sima, Gallego, Soledad, Ferrari, Andrea, and Jenney, Meriel

Published
2022
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34. Non-parameningeal head and neck rhabdomyosarcoma in children, adolescents, and young adults: Experience of the European paediatric Soft tissue sarcoma Study Group (EpSSG) – RMS2005 study

Author

Glosli, Heidi, primary, Bisogno, Gianni, additional, Kelsey, Anna, additional, Chisholm, Julia C., additional, Gaze, Mark, additional, Kolb, Frederic, additional, McHugh, Kieran, additional, Shipley, Janet, additional, Gallego, Soledad, additional, Merks, Johannes H.M., additional, Smeele, Ludi E., additional, Mandeville, Henry, additional, Ferrari, Andrea, additional, Minard-Colin, Veronique, additional, Corradini, Nadege, additional, Jenney, Meriel, additional, Zanetti, Ilaria, additional, De Salvo, Gian L., additional, and Orbach, Daniel, additional

Published
2021
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37. Immune reconstitution in children following chemotherapy for Acute Lymphoblastic Leukaemia

Author

Williams, Anthony, Bate, Jessica, Brooks, Rachel, Clarke, Stuart, Dixon, Elizabeth, Faust, Saul, Chisholm, Julia C., Galanopoulou, Angeliki, Heath, Paul, Maishman, Thomas, Mapstone, Susan, Patel, Soonie R., Vora, Ajay, Wilding, Sam, and Gray, Juliet

Abstract

Although survival rates for paediatric Acute Lymphoblastic Leukaemia are now excellent, this is at the expense of prolonged chemotherapy regimens. We report the long-term immune effects in children treated according to the UK Medical Research Council UKALL 2003 protocol. Peripheral blood lymphocyte subsets and immunoglobulin levels were studied in 116 participants, at 6 time points, during and for 18-months following treatment, with 30-39 patients analysed at each time point.Total lymphocytes were reduced during maintenance chemotherapy and remained low 18-months following treatment completion. CD4 T cells remained significantly reduced 18-months after treatment, but CD8 cells and natural killer cells recovered to normal values. The fall in naïve B cell numbers during maintenance was most marked, but numbers recovered rapidly after cessation of treatment. Memory B-cells, particularly non class-switched memory B-cells, remained below normal levels 18 months following treatment. All immunoglobulin subclasses were reduced during treatment compared to normal values, with IgM levels most affected. This study demonstrates that immune reconstitution differs between lymphocyte compartments. Although total B-cell numbers recover rapidly, disruption of memory/naïve balance persists and T cell compartment persist at 18-months. This highlights the impact of modern chemotherapy regimens on immunity, and thus infectious susceptibility and response to immunisation. Although survival rates for paediatric Acute Lymphoblastic Leukaemia are now excellent, this is at the expense of prolonged chemotherapy regimens. We report the long-term immune effects in children treated according to the UK Medical Research Council UKALL 2003 protocol. Peripheral blood lymphocyte subsets and immunoglobulin levels were studied in 116 participants, at 6 time points, during and for 18-months following treatment, with 30-39 patients analysed at each time point.Total lymphocytes were reduced during maintenance chemotherapy and remained low 18-months following treatment completion. CD4 T cells remained significantly reduced 18-months after treatment, but CD8 cells and natural killer cells recovered to normal values. The fall in naïve B cell numbers during maintenance was most marked, but numbers recovered rapidly after cessation of treatment. Memory B-cells, particularly non class-switched memory B-cells, remained below normal levels 18 months following treatment. All immunoglobulin subclasses were reduced during treatment compared to normal values, with IgM levels most affected. This study demonstrates that immune reconstitution differs between lymphocyte compartments. Although total B-cell numbers recover rapidly, disruption of memory/naïve balance persists and T cell compartment persist at 18-months. This highlights the impact of modern chemotherapy regimen

Published
2020

43. Phase II results from a phase I/II study to assess the safety and efficacy of weekly nab-paclitaxel in paediatric patients with recurrent or refractory solid tumours: A collaboration with the European Innovative Therapies for Children with Cancer Network

Author

Amoroso, Loredana, primary, Castel, Victoria, additional, Bisogno, Gianni, additional, Casanova, Michela, additional, Marquez-Vega, Catalina, additional, Chisholm, Julia C., additional, Doz, François, additional, Moreno, Lucas, additional, Ruggiero, Antonio, additional, Gerber, Nicolas U., additional, Fagioli, Franca, additional, Hingorani, Pooja, additional, Melcón, Soledad G., additional, Slepetis, Ruta, additional, Chen, Nianhang, additional, le Bruchec, Yvan, additional, Simcock, Mathew, additional, and Vassal, Gilles, additional

Published
2020
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44. Outcomes of metastatic non-rhabdomyosarcoma soft tissue sarcomas (NRSTS) treated within the BERNIE study: a randomised, phase II study evaluating the addition of bevacizumab to chemotherapy

Author

Ferrari, Andrea, primary, Merks, Johannes H.M., additional, Chisholm, Julia C., additional, Orbach, Daniel, additional, Brennan, Bernadette, additional, Gallego, Soledad, additional, van Noesel, Max M., additional, McHugh, Kieran, additional, van Rijn, Rick R., additional, Gaze, Mark N., additional, Martelli, Helene, additional, Bergeron, Christophe, additional, Corradini, Nadege, additional, Minard-Colin, Veronique, additional, Bisogno, Gianni, additional, Geoerger, Birgit, additional, Caron, Hubert N., additional, De Salvo, Gian Luca, additional, and Casanova, Michela, additional

Published
2020
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48. Psychosocial well-being of long-term survivors of pediatric head-neck rhabdomyosarcoma

Author

Vaarwerk, Bas, Schoot, Reineke A, Maurice-Stam, Heleen, Slater, Olga, Hartley, Benjamin, Saeed, Peerooz, Gajdosova, Eva, van den Brekel, Michiel W, Balm, Alfons J M, Hol, Marinka L F, van Jaarsveld, Stefanie, Kremer, Leontien C M, Ronckers, Cecile M, Mandeville, Henry C, Pieters, Bradley R, Gaze, Mark N, Davila Fajardo, Raquel, Strackee, Simon D, Dunaway, David, Smeele, Ludi E, Chisholm, Julia C, Caron, Huib N, Grootenhuis, Martha A, Merks, Johannes H M, Vaarwerk, Bas, Schoot, Reineke A, Maurice-Stam, Heleen, Slater, Olga, Hartley, Benjamin, Saeed, Peerooz, Gajdosova, Eva, van den Brekel, Michiel W, Balm, Alfons J M, Hol, Marinka L F, van Jaarsveld, Stefanie, Kremer, Leontien C M, Ronckers, Cecile M, Mandeville, Henry C, Pieters, Bradley R, Gaze, Mark N, Davila Fajardo, Raquel, Strackee, Simon D, Dunaway, David, Smeele, Ludi E, Chisholm, Julia C, Caron, Huib N, Grootenhuis, Martha A, and Merks, Johannes H M

Published
2019

49. A Perspective on Polo-Like Kinase-1 Inhibition for the Treatment of Rhabdomyosarcomas

Author

Gatz, Susanne A, Aladowicz, Ewa, Casanova, Michela, Chisholm, Julia C, Kearns, Pamela R, Fulda, Simone, Geoerger, Birgit, Schäfer, Beat W, Shipley, Janet M, Gatz, Susanne A, Aladowicz, Ewa, Casanova, Michela, Chisholm, Julia C, Kearns, Pamela R, Fulda, Simone, Geoerger, Birgit, Schäfer, Beat W, and Shipley, Janet M

Abstract

Rhabdomyosarcomas are the most common pediatric soft tissue sarcoma and are a major cause of death from cancer in young patients requiring new treatment options to improve outcomes. High-risk patients include those with metastatic or relapsed disease and tumors with PAX3-FOXO1 fusion genes that encode a potent transcription factor that drives tumourigenesis through transcriptional reprogramming. Polo-Like Kinase-1 (PLK1) is a serine/threonine kinase that phosphorylates a wide range of target substrates and alters their activity. PLK1 functions as a pleiotropic master regulator of mitosis and regulates DNA replication after stress. Taken together with high levels of expression that correlate with poor outcomes in many cancers, including rhabdomyosarcomas, it is an attractive therapeutic target. This is supported in rhabdomyosarcoma models by characterization of molecular and phenotypic effects of reducing and inhibiting PLK1, including changes to the PAX3-FOXO1 fusion protein. However, as tumor re-growth has been observed, combination strategies are required. Here we review preclinical evidence and consider biological rationale for PLK1 inhibition in combination with drugs that promote apoptosis, interfere with activity of PAX3-FOXO1 and are synergistic with microtubule-destabilizing drugs such as vincristine. The preclinical effects of low doses of the PLK1 inhibitor volasertib in combination with vincristine, which is widely used in rhabdomyosarcoma treatment, show particular promise in light of recent clinical data in the pediatric setting that support achievable volasertib doses predicted to be effective. Further development of novel therapeutic strategies including PLK1 inhibition may ultimately benefit young patients with rhabdomyosarcoma and other cancers.

Published
2019

50. Psychosocial well-being of long-term survivors of pediatric head-neck rhabdomyosarcoma

Author

MS Radiotherapie, Cancer, Vaarwerk, Bas, Schoot, Reineke A, Maurice-Stam, Heleen, Slater, Olga, Hartley, Benjamin, Saeed, Peerooz, Gajdosova, Eva, van den Brekel, Michiel W, Balm, Alfons J M, Hol, Marinka L F, van Jaarsveld, Stefanie, Kremer, Leontien C M, Ronckers, Cecile M, Mandeville, Henry C, Pieters, Bradley R, Gaze, Mark N, Davila Fajardo, Raquel, Strackee, Simon D, Dunaway, David, Smeele, Ludi E, Chisholm, Julia C, Caron, Huib N, Grootenhuis, Martha A, Merks, Johannes H M, MS Radiotherapie, Cancer, Vaarwerk, Bas, Schoot, Reineke A, Maurice-Stam, Heleen, Slater, Olga, Hartley, Benjamin, Saeed, Peerooz, Gajdosova, Eva, van den Brekel, Michiel W, Balm, Alfons J M, Hol, Marinka L F, van Jaarsveld, Stefanie, Kremer, Leontien C M, Ronckers, Cecile M, Mandeville, Henry C, Pieters, Bradley R, Gaze, Mark N, Davila Fajardo, Raquel, Strackee, Simon D, Dunaway, David, Smeele, Ludi E, Chisholm, Julia C, Caron, Huib N, Grootenhuis, Martha A, and Merks, Johannes H M

Published
2019

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