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Ewings sarcoma

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https://www.readbyqxmd.com/read/28338569/case-report-immune-mediated-complete-response-in-a-patient-with-recurrent-advanced-ewing-sarcoma-ews-after-vigil-immunotherapy
#1
Maurizio Ghisoli, McCarley Rutledge, Philip J Stephens, Robert Mennel, Minal Barve, Meghan Manley, Bahram R Oliai, Kathleen M Murphy, Luisa Manning, Belen Gutierrez, Priyanka Rangadass, Ashli Walker, Zhaohui Wang, Donald Rao, Ned Adams, Gladice Wallraven, Neil Senzer, John Nemunaitis
Ewing sarcoma is a highly resistant disease with a <10% chance of survival at 5 years after failure of frontline chemotherapy. This is a case report of an Ewing sarcoma patient with metastatic disease recurrence <2 years after standard chemotherapy/radiation who achieved a durable and sustained complete response after 2 series of treatments with Vigil (GMCSF/bi-shRNA furin DNA autologous tumor immunotherapy) serially manufactured from first and second recurrences with ELISPOT assay correlation. Results support justification of further testing of Vigil with ELISPOT assay as a biomarker to assess level of immune response and correlation with disease control...
March 23, 2017: Journal of Pediatric Hematology/oncology
https://www.readbyqxmd.com/read/28337592/head-and-neck-round-cell-sarcomas-a-comparative-clinicopathologic-analysis-of-2-molecular-subsets-ewing-and-cic-rearranged-sarcomas
#2
Adepitan A Owosho, Cherry L Estilo, Joseph M Huryn, Lei Zhang, Christopher D M Fletcher, Cristina R Antonescu
CIC-rearranged sarcoma (CRS) is a relatively new entity defined by its pathognomonic genetic signature and undifferentiated round cell phenotype, initially grouped together with the 'Ewing sarcoma-like tumors'. However, increasing data suggest that these tumors should be regarded as a stand-alone pathologic entity. We conducted a clinicopathologic analysis on molecularly conformed Ewing sarcoma (ES) and CRS arising in the head and neck (HN) and compared to a well characterized cohort of ES and CRS from other locations...
March 23, 2017: Head and Neck Pathology
https://www.readbyqxmd.com/read/28336564/identification-of-a-novel-syk-c-myc-malat1-signaling-pathway-and-its-potential-therapeutic-value-in-ewing-sarcoma
#3
Haibo Sun, De-Chen Lin, Qi Cao, Brendan Pang, David D Gae, Victor Km Lee, Huey Jin Lim, Ngan Doan, Jonathan W Said, Sigal Gery, Marilynn Chow, Anand Mayakonda, Charles Forscher, Jeffrey W Tyner, H Phillip Koeffler
PURPOSE: Ewing Sarcoma (EWS) is a devastating soft tissue sarcoma affecting predominantly young individuals. Tyrosine kinases (TKs) and associated pathways are continuously activated in many malignancies including EWS; these enzymes provide candidate therapeutic targets. EXPERIMENTAL DESIGN: Two high-throughput screens (a siRNA library and a small-molecule inhibitor library) were performed in EWS cells to establish candidate targets. Spleen tyrosine kinase (SYK) phosphorylation was assessed in EWS patients and cell lines...
March 23, 2017: Clinical Cancer Research: An Official Journal of the American Association for Cancer Research
https://www.readbyqxmd.com/read/28336328/bone-healing-is-achievable-despite-extensive-wound-dehiscence-and-wide-plate-and-screws-exposure-in-children
#4
Elias Melhem, Wael Bayoud, Ismat Ghanem
BACKGROUD: Extensive wound dehiscence and wide plate and screw exposure in the early weeks following orthopaedic surgery in children is usually managed either by extensive debridement, lavage and secondary closure or hardware removal and external fixation. PATIENTS: Three children with LCP plate and screws exposure were managed by simple repetitive debridement and local wound care without any IV antibiotics, nor secondary closure or hardware removal. All three cases occurred in the tibia, one following tibial osteotomy in lateral hemimelia with a long history of previous surgeries, one following wide excision of a tibial Ewing sarcoma with chemo and radiotherapy, and the third following wide excision of a 12cm necrotic tibial segment due to chronic osteomyelitis...
March 20, 2017: Orthopaedics & Traumatology, Surgery & Research: OTSR
https://www.readbyqxmd.com/read/28321815/immune-therapy-for-sarcomas
#5
Peter M Anderson
Absolute lymphocyte count (ALC) recovery rapidly occurring at 14 days after start of chemotherapy for osteosarcoma and Ewing sarcoma is a good prognostic factor. Conversely, lymphopenia is associated with significantly decreased sarcoma survival. Clearly, the immune system can contribute towards better survival from sarcoma. This chapter will describe treatment and host factors that influence immune function and how effective local control and systemic interventions of sarcoma therapy can cause inflammation and/or immune suppression but are currently the standard of care...
2017: Advances in Experimental Medicine and Biology
https://www.readbyqxmd.com/read/28319320/the-endochondral-bone-protein-chm1-sustains-an-undifferentiated-invasive-phenotype-promoting-lung-metastasis-in-ewing-sarcoma
#6
Kristina von Heyking, Julia Calzada-Wack, Stefanie Göllner, Frauke Neff, Oxana Schmidt, Tim Hensel, David Schirmer, Annette Fasan, Irene Esposito, Carsten Müller-Tidow, Poul H Sorensen, Stefan Burdach, Günther H S Richter
Ewing sarcomas (ES) are highly malignant, osteolytic bone or soft tissue tumors, which are characterized by EWS-ETS translocations and early metastasis to lung and bone. In this study, we investigated the role of the BRICHOS chaperone domain-containing endochondral bone protein chondromodulin I (CHM1) in ES pathogenesis. CHM1 is significantly over-expressed in ES, and chromosome immunoprecipitation (ChIP) data demonstrate CHM1 to be directly bound by an EWS-ETS translocation, EWS-FLI1. Using RNA interference we observed that CHM1 promoted chondrogenic differentiation capacity of ES cells but decreased the expression of osteolytic genes such as HIF1A, IL6, JAG1 and VEGF...
March 20, 2017: Molecular Oncology
https://www.readbyqxmd.com/read/28319067/the-histone-demethylase-kdm3a-and-its-downstream-target-mcam-promote-ewing-sarcoma-cell-migration-and-metastasis
#7
M Sechler, J K Parrish, D K Birks, P Jedlicka
Ewing Sarcoma is the second most common solid pediatric malignant neoplasm of bone and soft tissue. Driven by EWS/Ets, or rarely variant, oncogenic fusions, Ewing Sarcoma is a biologically and clinically aggressive disease with a high propensity for metastasis. However, the mechanisms underpinning Ewing Sarcoma metastasis are currently not well understood. In the present study, we identify and characterize a novel metastasis-promotional pathway in Ewing Sarcoma, involving the histone demethylase KDM3A, previously identified by our laboratory as a new cancer-promoting gene in this disease...
March 20, 2017: Oncogene
https://www.readbyqxmd.com/read/28296680/primitive-neuroectodermal-tumors-of-the-female-genital-tract-a-morphologic-immunohistochemical-and-molecular-study-of-19-cases
#8
Sarah Chiang, Matija Snuderl, Sakiko Kojiro-Sanada, Ariadna Quer Pi-Sunyer, Dean Daya, Tohru Hayashi, Luisanna Bosincu, Fumihiro Ogawa, Andrew E Rosenberg, Lars-Christian Horn, Lu Wang, A John Iafrate, Esther Oliva
Primary primitive neuroectodermal tumor (PNET) of the female genital tract is rare, and its proper classification remains unclear. The clinical, histologic, and immunophenotypic features as well as EWSR1 rearrangement status of 19 gynecologic PNETs, including 10 ovarian, 8 uterine, and 1 vulvar tumors, are herein reported. Patient age ranged from 12 to 68 years, with a median age of 20 and 51 years among those with ovarian and uterine PNETs, respectively. Morphologic features of central nervous system (CNS) tumors were seen in 15 PNETs, including 9 medulloblastomas, 3 ependymomas, 2 medulloepitheliomas, and 1 glioblastoma, consistent with central PNET...
March 14, 2017: American Journal of Surgical Pathology
https://www.readbyqxmd.com/read/28296559/the-irony-of-highly-effective-bacterial-therapy-of-a-patient-derived-orthotopic-xenograft-pdox-model-of-ewing-s-sarcoma-which-was-blocked-by-ewing-himself-80%C3%A2-years-ago
#9
Takashi Murakami, Tasuku Kiyuna, Kei Kawaguchi, Kentaro Igarashi, Arun S Singh, Yukihiko Hiroshima, Yong Zhang, Ming Zhao, Kentaro Miyake, Scott D Nelson, Sarah M Dry, Yunfeng Li, Jonathan C DeLong, Thinzar M Lwin, Takashi Chishima, Kuniya Tanaka, Michael Bouvet, Itaru Endo, Fritz C Eilber, Robert M Hoffman
William B. Coley developed bacterial therapy of cancer more than 100 years ago and had clinical success. James Ewing, a very famous cancer pathologist for whom the Ewing sarcoma is named, was Coley's boss at Memorial Hospital in New York and terminated Coley's bacterial therapy of cancer. A tumor from a patient with soft-tissue Ewing's sarcoma, who failed doxorubicin (DOX) therapy, was previously implanted in nude mice to establish a patient-derived orthotopic xenograft (PDOX) model. In the present study, the Ewing's sarcoma PDOX was treated with tumor-targeting S...
March 15, 2017: Cell Cycle
https://www.readbyqxmd.com/read/28293503/near-complete-humerus-reconstruction-in-the-pediatric-patient-with-vascularized-free-fibula-transfer
#10
John Shuck, Benjamin C Wood, Christopher Zarella, Albert K Oh, Robert M Henshaw, Gary F Rogers
Vascularized free fibula transfer remains the gold standard for reconstruction of large segmental diaphyseal defects of the upper extremity. In the pediatric patient, before skeletal maturity, free fibula transfer with the fibular head provides an active physis for growth and an articular interface for glenohumeral joint reconstruction. Clinical and cadaveric studies have demonstrated that the vascular supply to the fibular head originates, in most cases, from the anterior tibial system. However, anatomical variation exists, and we report a case in which a vascularized fibula autograft including the physis was transferred on the peroneal artery in a 5-year-old patient with Ewing's sarcoma...
December 2016: Plastic and Reconstructive Surgery. Global Open
https://www.readbyqxmd.com/read/28289810/non-odontogenic-tumors-of-the-facial-bones-in-children-and-adolescents-role-of-multiparametric-imaging
#11
REVIEW
Minerva Becker, Salvatore Stefanelli, Anne-Laure Rougemont, Pierre Alexandre Poletti, Laura Merlini
Tumors of the pediatric facial skeleton represent a major challenge in clinical practice because they can lead to functional impairment, facial deformation, and long-term disfigurement. Their treatment often requires a multidisciplinary approach, and radiologists play a pivotal role in the diagnosis and management of these lesions. Although rare, pediatric tumors arising in the facial bones comprise a wide spectrum of benign and malignant lesions of osteogenic, fibrogenic, hematopoietic, neurogenic, or epithelial origin...
March 13, 2017: Neuroradiology
https://www.readbyqxmd.com/read/28287344/ewing-s-sarcoma%C3%A2-of-the-cranial-vault-a-case-report
#12
Jihene Feki, Zeineb Guermazi, Brahim Kammoun, Afef Khanfir, Nabil Toumi, Tahiya Boudawara, Zaher Boudawara, Jamel Daoud, Mounir Frikha
Ewing's sarcoma is a malignant tumor that mainly affects young patients. It represents 10% of primary malignant tumors of the bone and 3% of malignant tumors of the child. Cranial localization is extremely rare representing less than 1% of all the localizations. We report a case of a 10-year-old girl who presented with an intracranial hypertension syndrome with left parietal mass of progressive installation. The X-ray skull showed a lytic lesion with irregular margins involving the left parietal bone. Brain magnetic resonance imaging revealed extensive parietal bone destruction involving both the inner and outer tables...
March 13, 2017: Acta Clinica Belgica
https://www.readbyqxmd.com/read/28278156/a-new-mode-of-dna-binding-distinguishes-capicua-from-other-hmg-box-factors-and-explains-its-mutation-patterns-in-cancer
#13
Marta Forés, Lucía Simón-Carrasco, Leiore Ajuria, Núria Samper, Sergio González-Crespo, Matthias Drosten, Mariano Barbacid, Gerardo Jiménez
HMG-box proteins, including Sox/SRY (Sox) and TCF/LEF1 (TCF) family members, bind DNA via their HMG-box. This binding, however, is relatively weak and both Sox and TCF factors employ distinct mechanisms for enhancing their affinity and specificity for DNA. Here we report that Capicua (CIC), an HMG-box transcriptional repressor involved in Ras/MAPK signaling and cancer progression, employs an additional distinct mode of DNA binding that enables selective recognition of its targets. We find that, contrary to previous assumptions, the HMG-box of CIC does not bind DNA alone but instead requires a distant motif (referred to as C1) present at the C-terminus of all CIC proteins...
March 2017: PLoS Genetics
https://www.readbyqxmd.com/read/28272685/therapy-induced-neural-differentiation-in-ewing-s-sarcoma-a-case-report-and-review-of-the-literature
#14
Kıvılcım Eren Erdoğan, Mehmet Ali Deveci, Zeynep Ruken Hakkoymaz, Gülfiliz Gönlüşen
Ewing's sarcoma (ES) is a small round cell tumor of adolescents or young adults that usually arises in the deep soft tissues of the extremities. The tumor cells have uniform round nuclei, fine powdery chromatin and indistinct nucleoli. CD99 (O13) is a product of the MIC 2 gene that is highly sensitive to ES but not specific. A panel of markers should be used for the differential diagnosis of small round cell tumors because nearly all others, on occasion, show membranous staining for CD99. One of the defining feature of ES is the presence of 22q12 gene rearrangement...
February 4, 2017: Türk Patoloji Dergisi
https://www.readbyqxmd.com/read/28272661/primary-paediatric-renal-primitive-neuroectodermal-tumor-a-case-report-and-review-of-the-literature
#15
Binny Khandakar, Moumita Maiti, Soumit Dey, Prasenjit Sen Ray, Palas Bhattac Haryya, Ranu Sarkar
Ewing sarcoma/primitive neuroectodermal tumor (PNET) is a high-grade malignant neoplasm commonly affecting bones of the thoracic region. Primary Ewing sarcoma/PNET of the kidney is exceptional; it commonly affects young adults and is rarely reported in children. Here we describe a case of renal PNET in a 10-year-old girl who presented at our institute with haematuria and abdominal lump. Computer tomography scan revealed a huge mass in the right kidney. Computer tomography-guided aspiration from the mass showed cytomorphological features of a small blue round cell tumor...
February 4, 2017: Türk Patoloji Dergisi
https://www.readbyqxmd.com/read/28271180/metal-wear-induced-pseudotumour-following-an-endoprosthetic-knee-replacement-for-ewing-sarcoma
#16
Richard Craig, Marianna Vlychou, Catherine L McCarthy, Christopher L M H Gibbons, N A Athanasou
Pseudotumours are well recognised as a complication of metal-on-metal hip arthroplasties and are thought to develop on the basis of an innate and adaptive immune response to cobalt-chrome (Co-Cr) wear particles. We report a case of a large pseudotumour that developed following a knee endoprosthetic replacement (EPR) undertaken for Ewing sarcoma. The lesion contained necrotic and degenerate connective tissue in which there were numerous scattered metal wear-containing macrophages, eosinophil polymorphs, lymphocytes, plasma cells and aseptic lymphocyte-dominated vascular-associated lesion-like lymphoid aggregates...
March 7, 2017: Skeletal Radiology
https://www.readbyqxmd.com/read/28266080/efficacy-comparison-of-six-chemotherapeutic-combinations-for-osteosarcoma-and-ewing-s-sarcoma-treatment-a-network-meta-analysis
#17
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
https://www.readbyqxmd.com/read/28262274/sinonasal-adamantinoma-like-ewing-sarcoma-a-case-report
#18
Borislav A Alexiev, Yanki Tumer, Justin A Bishop
We describe the case of a sinonasal adamantinoma-like Ewing sarcoma in a 41-year-old male. Histologically, the tumor exhibited distinctive areas of nested growth pattern with prominent stromal fibrosis and metaplastic bone formation. The tumor cells were small and uniform with minimal amount of pale eosinophilic to clear cytoplasm and round or oval nuclei with finely dispersed chromatin and small nucleoli. Approximately 20% of the tumor parenchyma comprised of small clusters of basaloid cells within an osteofibrous background resembling adamantinoma...
December 2, 2016: Pathology, Research and Practice
https://www.readbyqxmd.com/read/28258912/risk-stratification-and-pattern-of-cardiotoxicity-in-pediatric-ewing-sarcoma
#19
Emad Moussa, Manal Zamzam, Ahmed Kamel, Zeinab Salah, Iman Attia, Lina Gaber, Ranin Soliman, Sameera Ezzat
INTRODUCTION: Anthracycline chemotherapy contributes to improved outcomes in Ewing sarcoma; however, the most feared complication is cardiotoxicity. Echocardiograms were routinely used to monitor cardiac function after anthracycline treatment. Nevertheless, indices chosen to assess cardiac toxicity vary significantly among different centers, and no uniform protocol has been accepted as ideal. METHODS: This retrospective study included children with Ewing sarcoma treated at Children's Cancer Hospital Egypt over 4years...
February 28, 2017: Journal of the Egyptian National Cancer Institute
https://www.readbyqxmd.com/read/28255420/complete-resection-of-a-primitive-neuroectodermal-tumour-arising-in-the-bladder-of-a-31-year-old-female-after-neoadjuvant-chemotherapy
#20
Cameron J Lam, Bobby Shayegan
Primitive neuroectodermal tumours (PNET) that arise in the urinary bladder are an extremely rare occurrence. Very few cases have been reported so far in the literature1-13 and we report another case here in a 31-year-old-female. The patient presented with polyuria, gross hematuria, followed by development of anuria, and was discovered to have a 9.4 cm mass arising in the posterolateral aspect of the bladder. Histologically, the tumour showed small, round, blue cells. Further analysis using break-apart fluorescent in situ hybridization (FISH) revealed non-random chromosomal translocations of the ews gene suggestive of Ewing sarcoma (ES)/PNET...
July 2016: Canadian Urological Association Journal, Journal de L'Association des Urologues du Canada
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