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Ewing relapse

Danielle Novetsky Friedman, Katherine Chastain, Joanne F Chou, Chaya S Moskowitz, Roberto Adsuar, Leonard H Wexler, Alexander J Chou, Amelia DeRosa, Joanne Candela, Heather Magnan, Shawn Pun, Tamara Kahan, Suzanne L Wolden, Paul A Meyers, Kevin C Oeffinger
BACKGROUND: Children, adolescents, and young adults treated for Ewing sarcoma (ES) are at risk for disease-related and treatment-related complications. We aimed to describe early and late overall mortality, cause-specific mortality, and key adverse health outcomes in a large, single-institutional cohort of patients with ES. METHODS: Patients with ES diagnosed at age less than 40 years and treated at Memorial Sloan Kettering between 1974 and 2012 were included. Overall survival was estimated using Kaplan-Meier methods...
April 18, 2017: Pediatric Blood & Cancer
Lora E Burke, Saul Shiffman, Edvin Music, Mindi A Styn, Andrea Kriska, Asim Smailagic, Daniel Siewiorek, Linda J Ewing, Eileen Chasens, Brian French, Juliet Mancino, Dara Mendez, Patrick Strollo, Stephen L Rathbun
BACKGROUND: Ecological momentary assessment (EMA) assesses individuals' current experiences, behaviors, and moods as they occur in real time and in their natural environment. EMA studies, particularly those of longer duration, are complex and require an infrastructure to support the data flow and monitoring of EMA completion. OBJECTIVE: Our objective is to provide a practical guide to developing and implementing an EMA study, with a focus on the methods and logistics of conducting such a study...
March 15, 2017: Journal of Medical Internet Research
Tao Zhang, Song Zhang, Feifei Yang, Lili Wang, Sigang Zhu, Bing Qiu, Shunhua Li, Zhongliang Deng
This study aimed to address the insufficiency of traditional meta-analysis and provide improved guidelines for the clinical practice of osteosarcoma treatment. The heterogeneity of the fixed-effect model was calculated, and when necessary, a random-effect model was adopted. Furthermore, the direct and indirect evidence was pooled together and exhibited in the forest plot and slash table. The surface under the cumulative ranking curve (SUCRA) value was also measured to rank each intervention. Finally heat plot was introduced to demonstrate the contribution of each intervention and the inconsistency between direct and indirect comparisons...
March 7, 2017: Journal of Cellular Biochemistry
Christopher Kuo, Paul M Kent, Antonio D Logan, Karen B Tamulonis, Kristen L Dalton, Marta Batus, Karen Fernandez, Rebecca E Mcfall
BACKGROUND: Adolescent and young adult (AYA) patients with very high risk sarcomas have poor outcomes and are in need of novel therapies. PROCEDURE: From January 2005 to February 2016, we retrospectively identified all AYA patients with relapsed or metastatic high-grade sarcomas, who were treated with at least one cycle of docetaxel (T), bevacizumab (A), and gemcitabine (G) (TAG ; T = 100 mg/m(2) Day 8, A = 15 mg/kg Day 1, G = 1,000 mg/m(2) Days 1 and 8). RESULTS: Fourteen patients, median age of 20 (15-30), received a total of 80 cycles of TAG, and were followed for a median of 83 months...
April 2017: Pediatric Blood & Cancer
M N Hayes, D M Langenau
Sarcoma is a type of cancer affecting connective, supportive, or soft tissue of mesenchymal origin. Despite rare incidence in adults (<1%), over 15% of pediatric cancers are sarcoma. Sadly, both adults and children with relapsed or metastatic disease have devastatingly high rates of mortality. Current treatment options for sarcoma include surgery, radiation, and/or chemotherapy; however, significant limitations exist with respect to the efficacy of these strategies. Strong impetus has been placed on the development of novel therapies and preclinical models for uncovering mechanisms involved in the development, progression, and therapy resistance of sarcoma...
2017: Methods in Cell Biology
Antonino Di Franco, Mario Gaudino, Jonathan W Weinsaft, Shawn C Pun, Navneet Narula, Saadat A Khan, Zahra M Malik, Lucas B Ohmes, Nickolaos J Skubas, Leonard N Girardi
A high suspicion for relapsed metastatic disease must arise when an intracardiac mass is detected in a patient with a recent history of Ewing sarcoma. Nevertheless, the scenario may eventually turn out to be much more complex than expected, and the possibility that the intracardiac tumor may instead be a "second" primary sarcoma, although extremely rare, should also be considered. We describe the first case of concomitant diagnosis of Ewing sarcoma and low-grade myxoid spindle cell sarcoma in the same young patient...
February 2017: Annals of Thoracic Surgery
Jessica H Cheng, Leslie Y Chiang, Dennis John Kuo
Complementary and alternative medicine (CAM) therapies are commonly incorporated into the care of patients with paediatric cancer. Many modalities are safe and effective during cancer treatment and have proved beneficial for symptom relief and quality of life. However, situations where alternative therapy is provided without allopathic medical care supportive care resources can pose a safety risk to patients. This report describes the case of a 16-year-old Chinese girl with metastatic Ewing sarcoma who sought treatment with alternative treatment in Mexico...
January 6, 2017: BMJ Case Reports
Marcella Martinelli, Alessandro Parra, Luca Scapoli, Paola De Sanctis, Valentina Chiadini, Claudia Hattinger, Piero Picci, Cinzia Zucchini, Katia Scotlandi
Ewing sarcoma (EWS), the second most common primary bone tumor in pediatric age, is known for its paucity of recurrent somatic abnormalities. Apart from the chimeric oncoprotein that derives from the fusion of EWS and FLI genes, recent genome-wide association studies have identified susceptibility variants near the EGR2 gene that regulate DNA binding of EWS-FLI. However, to induce transformation, EWS-FLI requires the presence of additional molecular events, including the expression of CD99, a cell surface molecule with critical relevance for the pathogenesis of EWS...
November 22, 2016: Oncotarget
Wei Wan, Yan Lou, Zhiqi Hu, Ting Wang, Jinsong Li, Yu Tang, Zhipeng Wu, Leqin Xu, Xinghai Yang, Dianwen Song, Jianru Xiao
Little information has been published in the literature regarding survival outcomes of patients with Ewing's sarcoma family tumors (ESFTs) of the spine. The purpose of this study is to explore factors that may affect the prognosis of patients with non-metastatic spinal ESFTs. A retrospective analysis of survival outcomes was performed in patients with non-metastatic spinal ESFTs. Univariate and multivariate analyses were employed to identify prognostic factors for recurrence and survival. Recurrence-free survival (RFS) and overall survival (OS) were defined as the date of surgery to the date of local relapse and death...
January 2017: Journal of Neuro-oncology
Winston W Huh, Najat C Daw, Cynthia E Herzog, Mark F Munsell, Mary Frances McAleer, Valerae O Lewis
AIM: Few data exist regarding the clinical characteristics and outcome of young children with Ewing sarcoma family of tumors (ESFT). METHODS: We reviewed the records of ESFT patients at our institution younger than 10 years of age at diagnosis. RESULTS: Forty-two patients were identified. Median age was 6.4 years (range 0.6-9.5 years). Most patients had T2 (>5 cm) tumors (n = 31; 74%). Most common primary site was the extremity (n = 17; 41%)...
October 3, 2016: Pediatric Blood & Cancer
Kathleen I Pishas, Stephen L Lessnick
Ewing sarcoma is an aggressive, poorly differentiated neoplasm of solid bone that disproportionally afflicts the young. Despite intensive multi-modal therapy and valiant efforts, 70% of patients with relapsed and metastatic Ewing sarcoma will succumb to their disease. The persistent failure to improve overall survival for this subset of patients highlights the urgent need for rapid translation of novel therapeutic strategies. As Ewing sarcoma is associated with a paucity of mutations in readily targetable signal transduction pathways, targeting the key genetic aberration and master regulator of Ewing sarcoma, the EWS/ETS fusion, remains an important goal...
2016: F1000Research
Christine M Heske, Arnulfo Mendoza, Leah D Edessa, Joshua T Baumgart, Sunmin Lee, Jane Trepel, David A Proia, Len Neckers, Lee J Helman
Long-term survival in patients with metastatic, relapsed, or recurrent Ewing sarcoma and rhabdomyosarcoma is dismal. Irinotecan, a topoisomerase 1 inhibitor, has activity in these sarcomas, but due to poor bioavailability of its active metabolite (SN-38) has had limited clinical efficacy. In this study we have evaluated the efficacy and toxicity of STA-8666, a novel drug conjugate which uses an HSP90 inhibitor to facilitate intracellular, tumor-targeted delivery of the topoisomerase 1 inhibitor SN-38, thus preferentially delivering and concentrating SN-38 within tumor tissue...
October 4, 2016: Oncotarget
Monika Twardziok, Susann Kleinsimon, Jana Rolff, Sebastian Jäger, Angelika Eggert, Georg Seifert, Catharina I Delebinski
Ewing sarcoma is the second most common bone cancer in children and adolescents, with poor prognosis and outcome in ~70% of initial diagnoses and 10-15% of relapses. Hydrophobic triterpene acids and hydrophilic lectins and viscotoxins from European mistletoe (Viscum album L.) demonstrate anticancer properties, but have not yet been investigated for Ewing sarcoma. Commercial Viscum album L. extracts are aqueous, excluding the insoluble triterpenes. We recreated a total mistletoe effect by combining an aqueous extract (viscum) and a triterpene extract (TT) solubilized with cyclodextrins...
2016: PloS One
Camille Jacques, Lidia Rodriguez Calleja, Marc Baud'huin, Thibaut Quillard, Dominique Heymann, François Lamoureux, Benjamin Ory
Osteosarcoma and Ewing Sarcoma are the two most common types of Bone Sarcomas, principally localized at the long bones of the extremities and mainly affecting adolescents and young adults. Cisplatin is one of the current options in the therapeutic arsenal of drugs available to cure these aggressive cancers. Unfortunately, chemoresistance against this agent is still a major cause of patient relapse. Thus, a better understanding of the molecular pathways by which these drugs induce cancer cell death, together with a better delineation of the origins of chemoresistance are required to improve the success rate of current treatments...
August 23, 2016: Oncotarget
Uwe Thiel, Angela Wawer, Irene von Luettichau, Hans-Ulrich Bender, Franziska Blaeschke, Thomas G P Grunewald, Marc Steinborn, Barbara Röper, Halvard Bonig, Thomas Klingebiel, Peter Bader, Ewa Koscielniak, Michael Paulussen, Uta Dirksen, Heribert Juergens, Hans-Jochem Kolb, Stefan E G Burdach
PURPOSE: Advanced Ewing sarcomas have poor prognosis. They are defined by early relapse (<24 months after diagnosis) and/or by metastasis to multiple bones or bone marrow (BM). We analyzed risk factors, toxicity and survival in advanced Ewing sarcoma patients treated with the MetaEICESS vs. EICESS92 protocols. DESIGN: Of 44 patients, 18 patients were enrolled into two subsequent MetaEICESS protocols between 1992 and 2014, and compared to outcomes of 26 advanced Ewing sarcoma patients treated with EICESS 1992 between 1992 and 1996...
October 25, 2016: Oncotarget
Barrie Cohen, Michael Roth, Jonathan M Marron, Stacy W Gray, David S Geller, Bang Hoang, Richard Gorlick, Katherine A Janeway, Jonathan Gill
BACKGROUND: Patients with relapsed and refractory solid tumors have a poor prognosis. Recent advances in genomic technology have made it feasible to screen tumors for actionable mutations, with the anticipation that this may provide benefit to patients. METHODS: Pediatric oncologists were emailed an anonymous 34-question survey assessing their willingness to offer a rebiopsy to patients with relapsed disease for the purpose of tumor genomic profiling. They were presented with two scenarios evaluating morbidity and invasiveness of the procedures using the clinical examples of medulloblastoma and Ewing sarcoma...
December 2016: Annals of Surgical Oncology
Gonçalo Videira, Pedro Castro, Bítia Vieira, João Pedro Filipe, Rosa Santos, Elsa Azevedo, Maria José Sá, Pedro Abreu
BACKGROUND: MS-associated autonomic dysfunction (AD) in multiple sclerosis (MS) is poorly understood and the best method for its detection unestablished. We compared classical Ewing battery and newer methods as heart rate variability (HRV) and spontaneous baroreflex sensibility (BRS) to detect AD in MS and related them to central autonomic network (CAN) lesions. METHODS: We enrolled 20 relapsing-remitting MS patients, median age of 36 (interquartile range 32-46) years, disease duration of 5...
August 15, 2016: Journal of the Neurological Sciences
Elisabeth A Pedersen, Rajasree Menon, Kelly M Bailey, Dafydd G Thomas, Raelene A Van Noord, Jenny Tran, Hongwei Wang, Ping Ping Qu, Antje Hoering, Eric R Fearon, Rashmi Chugh, Elizabeth R Lawlor
Ewing sarcomas are characterized by the presence of EWS/ETS fusion genes in the absence of other recurrent genetic alterations and mechanisms of tumor heterogeneity that contribute to disease progression remain unclear. Mutations in the Wnt/β-catenin pathway are rare in Ewing sarcoma but the Wnt pathway modulator LGR5 is often highly expressed, suggesting a potential role for the axis in tumor pathogenesis. We evaluated β-catenin and LGR5 expression in Ewing sarcoma cell lines and tumors and noted marked intra- and inter-tumor heterogeneity...
September 1, 2016: Cancer Research
Peter M Anderson, Stefan S Bielack, Richard G Gorlick, Keith Skubitz, Najat C Daw, Cynthia E Herzog, Odd R Monge, Alvaro Lassaletta, Erica Boldrini, Zsuzanna Pápai, Joseph Rubino, Kumudu Pathiraja, Darcy A Hille, Mark Ayers, Siu-Long Yao, Michael Nebozhyn, Brian Lu, David Mauro
BACKGROUND: Robatumumab (19D12; MK-7454 otherwise known as SCH717454) is a fully human antibody that binds to and inhibits insulin-like growth factor receptor-1 (IGF-1R). This multiinstitutional study (P04720) determined the safety and clinical efficacy of robatumumab in three separate patient groups with resectable osteosarcoma metastases (Group 1), unresectable osteosarcoma metastases (Group 2), and Ewing sarcoma metastases (Group 3). PROCEDURE: Robatumumab infusions were administered every 2 weeks and were well tolerated with minimal toxicity...
October 2016: Pediatric Blood & Cancer
Hannah Fassel, Donald Tracy, Katie Louer, Giannoula Lakka Klement
Despite the advances in oncology, the survival of children with Ewing Sarcoma metastatic at diagnosis continues to be 27% 3-year event-free survival and 34% 3-year overall survival. In other words, 7 of 10 children die within 3 years of their initial diagnosis despite intense chemotherapy, local treatment (radiation/surgery), and/or high dose busulfan-melphalan and autologous stem-cell transplantation. A chief contributor to this morbidity and mortality is the difficulty eradicating the tumor using present therapeutic modalities...
October 2016: Journal of Pediatric Hematology/oncology
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