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Journal articles on the topic 'Sarcoma Ewing'

Author: Grafiati

Published: 4 June 2021

Last updated: 4 February 2022

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1

Ramaswamy, Anant, Bharat Rekhi, Sameer Bakhshi, Sachin Hingmire, and Manish Agarwal. "Indian data on bone and soft tissue sarcomas: A summary of published study results." South Asian Journal of Cancer 05, no. 03 (July 2016): 138–45. http://dx.doi.org/10.4103/2278-330x.187587.

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AbstractBone sarcomas are rare tumors, approximating 0.2% of all cancers, with osteosarcoma (OGS), chondrosarcoma, and Ewing sarcoma being the most common cancers in this subset. The formation of disease management groups/clinics focused on sarcomas has resulted in better understanding and management of these uncommon tumors. Multiple large-scale retrospective data from Tata Memorial Hospital (TMH) and All India Institute of Medical Sciences have reported outcomes comparable to Western data in the field of OGS and Ewing sarcoma, with interesting prognostic factors identified for further evaluation. Soft tissue sarcomas are a rare heterogeneous group of tumors, more than 50 different tumor entities. The common subtypes identified in India include Ewing sarcoma and synovial sarcoma. Valuable work regarding brachytherapy has been done by radiation oncologists from the TMH, especially in pediatric patients.

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2

Ulusan, S., Z. Koc, E. Tuba Canpolat, and T. Çolakoglu. "Radiological findings of primary retroperitoneal ewing sarcoma." Acta Radiologica 48, no. 7 (September 2007): 814–18. http://dx.doi.org/10.1080/02841850701408244.

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Ewing sarcomas are most commonly located in bone, while extraskeletal involvement of the retroperitoneum is extremely rare. We describe the radiologic and pathological findings in an adult patient with retroperitoneal extraskeletal Ewing sarcoma.

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3

Anderson, Nathaniel D., Richard de Borja, Matthew D. Young, Fabio Fuligni, Andrej Rosic, Nicola D. Roberts, Simon Hajjar, et al. "Rearrangement bursts generate canonical gene fusions in bone and soft tissue tumors." Science 361, no. 6405 (August 30, 2018): eaam8419. http://dx.doi.org/10.1126/science.aam8419.

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Sarcomas are cancers of the bone and soft tissue often defined by gene fusions. Ewing sarcoma involves fusions between EWSR1, a gene encoding an RNA binding protein, and E26 transformation-specific (ETS) transcription factors. We explored how and when EWSR1-ETS fusions arise by studying the whole genomes of Ewing sarcomas. In 52 of 124 (42%) of tumors, the fusion gene arises by a sudden burst of complex, loop-like rearrangements, a process called chromoplexy, rather than by simple reciprocal translocations. These loops always contained the disease-defining fusion at the center, but they disrupted multiple additional genes. The loops occurred preferentially in early replicating and transcriptionally active genomic regions. Similar loops forming canonical fusions were found in three other sarcoma types. Chromoplexy-generated fusions appear to be associated with an aggressive form of Ewing sarcoma. These loops arise early, giving rise to both primary and relapse Ewing sarcoma tumors, which can continue to evolve in parallel.

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4

Świtaj, Tomasz, and Paulina Jagodzińska-Mucha. "Ewing sarcoma." Oncology in Clinical Practice 14, no. 6 (March 15, 2019): 392–98. http://dx.doi.org/10.5603/ocp.2018.0052.

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5

Szabo, Brittany, and Justin Gambini. "Ewing Sarcoma." JBJS Journal of Orthopaedics for Physician Assistants 8, no. 2 (2020): e0004-e0004. http://dx.doi.org/10.2106/jbjs.jopa.20.00004.

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6

Mankin, Henry J. "Ewing sarcoma." Current Opinion in Orthopedics 11, no. 6 (December 2000): 479–85. http://dx.doi.org/10.1097/00001433-200012000-00010.

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7

Casado, M., E. Rollán, S. Guardado, N. Gascón, A. Mañas, M. Cabeza, and J. Pérez-Regadera. "Ewing sarcoma." Reports of Practical Oncology & Radiotherapy 18 (June 2013): S200. http://dx.doi.org/10.1016/j.rpor.2013.03.154.

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8

Ju, Hee Young. "Ewing Sarcoma." Clinical Pediatric Hematology-Oncology 26, no. 1 (April 30, 2019): 27–34. http://dx.doi.org/10.15264/cpho.2019.26.1.27.

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9

Choi, Eun-Young K., Jerad M. Gardner, David R. Lucas, Jonathan B. McHugh, and Rajiv M. Patel. "Ewing sarcoma." Seminars in Diagnostic Pathology 31, no. 1 (January 2014): 39–47. http://dx.doi.org/10.1053/j.semdp.2014.01.002.

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10

Ben-Ami, Tal, Elisha Waldman, Wygoda Marc, Michael Weintraub, Shoshana Revel-Vilk, and Iris Fried. "Ewing Sarcoma." Journal of Pediatric Hematology/Oncology 38, no. 1 (January 2016): 38–42. http://dx.doi.org/10.1097/mph.0000000000000456.

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11

Stark, Andreas M., Ivo Leuschner, H. Maximilian Mehdorn, and Alexander Claviez. "Ewing Sarcoma of the Posterior Fossa in an Adolescent Girl." Case Reports in Medicine 2014 (2014): 1–4. http://dx.doi.org/10.1155/2014/439830.

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Medulloblastoma, astrocytoma, and ependymoma represent the most common infratentorial tumors in childhood, while Ewing sarcomas in that localization are extremely rare. A large left infratentorial space-occupying lesion was diagnosed in a 12-year-old girl with signs of increased intracranial pressure. Following total tumor resection, histological and molecular examination revealed Ewing sarcoma with rearrangedEWSR-1gene. The patient achieved complete remission following adjuvant chemotherapy and radiotherapy according to Euro-EWING 2008 treatment protocol. Intracranial Ewing sarcoma, although rare, should be an important differential diagnosis of intracranial tumors in childhood which requires aggressive multimodal treatment.

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12

Wu, S. Peter, Benjamin T. Cooper, Fang Bu, Christopher J. Bowman, J. Keith Killian, Jonathan Serrano, Shiyang Wang, et al. "DNA Methylation–Based Classifier for Accurate Molecular Diagnosis of Bone Sarcomas." JCO Precision Oncology, no. 1 (November 2017): 1–11. http://dx.doi.org/10.1200/po.17.00031.

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Purpose Pediatric sarcomas provide a unique diagnostic challenge. There is considerable morphologic overlap between entities, increasing the importance of molecular studies in the diagnosis, treatment, and identification of therapeutic targets. We developed and validated a genome-wide DNA methylation–based classifier to differentiate between osteosarcoma, Ewing sarcoma, and synovial sarcoma. Methods DNA methylation status of 482,421 CpG sites in 10 Ewing sarcoma, 11 synovial sarcoma, and 15 osteosarcoma samples were determined using the Illumina Infinium HumanMethylation450 array. We developed a random forest classifier trained from the 400 most differentially methylated CpG sites within the training set of 36 sarcoma samples. This classifier was validated on data drawn from The Cancer Genome Atlas synovial sarcoma, TARGET-Osteosarcoma, and a recently published series of Ewing sarcoma. Results Methylation profiling revealed three distinct patterns, each enriched with a single sarcoma subtype. Within the validation cohorts, all samples from The Cancer Genome Atlas were accurately classified as synovial sarcoma (10 of 10; sensitivity and specificity, 100%), all but one sample from TARGET-Osteosarcoma were classified as osteosarcoma (85 of 86; sensitivity, 98%; specificity, 100%), and 14 of 15 Ewing sarcoma samples were classified correctly (sensitivity, 93%; specificity, 100%). The single misclassified osteosarcoma sample demonstrated high EWSR1 and ETV1 expression on RNA sequencing, although no fusion was found on manual curation of the transcript sequence. Two additional clinical samples that were difficult to classify by morphology and molecular methods were classified as osteosarcoma; one had been suspected of being a synovial sarcoma and the other of being Ewing sarcoma on initial diagnosis. Conclusion Osteosarcoma, synovial sarcoma, and Ewing sarcoma have distinct epigenetic profiles. Our validated methylation-based classifier can be used to provide diagnostic assistance when histologic and standard techniques are inconclusive.

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13

Agrawal, Sonam, Vanita C. Rathod, Pankaj Kumar Agrawal, and Swati Saluja. "Case report on Ewing's sarcoma with review of literature." Asian Journal of Medical Sciences 6, no. 6 (May 11, 2015): 88–92. http://dx.doi.org/10.3126/ajms.v6i6.12283.

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Ewings sarcoma is a highly malignant primary mesenchymal neoplasm of bone,which was first reported in 1921 by Ewing. The exact cell of origin of Ewing Sarcoma is not known it is generally believed that the lesion arises from either the endothelial cells of the blood vessels within the bone or from the undifferentiated reticulo-endothelial cell. Primitive neuroectodermal tumors (PNETs) arises from mesenchymal stem cells that differentiates along neural cell lineage. Reports of Ewings sarcoma constitute about 10% of all malignant bone tumor. A case of Ewings sarcoma in 10 year old male patient is reported, who visited to Dept of Oral and Maxillo-facial Pathology in Rungta College of Dental Sciences And Research, with chief complaint of pain and swelling in right side of face since 1 month. Early diagnosis of this case helps in selecting the appropriate treatment procedure and to avoid further complication. DOI: http://dx.doi.org/10.3126/ajms.v6i6.12283Asian Journal of Medical Sciences Vol.6(6) 2015 88-92

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14

Tavakkoli, Montreh, and Lisa Mueller. "Cutaneous Ewing Sarcoma and Ewing Sarcoma of the Bone: Distinct Diseases." Case Reports in Oncology 11, no. 3 (November 12, 2018): 729–34. http://dx.doi.org/10.1159/000492667.

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Ewing sarcoma is an aggressive mesenchymal malignancy. It is the second most common bone tumor among children and adolescents and less commonly presents as a soft tissue or primary skin lesion. Cutaneous Ewing sarcoma has only been reported in case reports and case series. In this article, we describe a 12-year-old Hispanic female cured of localized, cutaneous Ewing sarcoma (pT1aN0M0) at the 40-month follow-up following surgical resection and adjuvant chemotherapy according to the COG AEWS1031 protocol for Ewing sarcoma of the bone. To our knowledge, this is the first article to provide a potential biological explanation for the differences in the prognosis of Ewing sarcoma of the bone, soft tissue, and skin.

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15

Ebrahimpour, Adel, Mohammadreza Chehrassan, Mehrdad Sadighi, Mehdi Azizmohammad Looha, Amin Karimi, Alireza Raeisi, Alireza Zali, and Mohammad Esmaeil Akbari. "Mobile Spine Osseous Sarcoma: Descriptive Epidemiological Analysis Based on a National Population-Based Study." Archives of Iranian Medicine 24, no. 8 (August 1, 2021): 629–35. http://dx.doi.org/10.34172/aim.2021.89.

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Background: Primary osseous sarcomas of the mobile spine are rare bony tumors. Ewing sarcoma, chondrosarcoma, chordoma and osteosarcoma constitute the majority of primary bone sarcomas of the spine; however, other rare sarcoma tumors may also affect the spine. In order to perform an epidemiological study of theses tumors, national registries may help to evaluate data for populations with similar characteristics. Methods: A population-based study was designed based on data from the Iran National Cancer Registry (INCR). All morphology codes (M-Code) of primary osseous sarcomas of the mobile spine (C-code 41.2) were derived and analyzed. Results: Among 186 patients with primary osseous sarcomas of the mobile spine, 67.2% were men and 32.8% were women. The median (IQR) age was 37.0 (20.0–59.0) years and the age-standardized incidence rate (ASIR) was 0.37 per million. The majority of cases of Ewing sarcoma (29.5%) were observed in the age group 20–25 years. Among male patients with chondrosarcoma, the median age was 39.0 (30.0–50.0), while females showed a median age of 56.0 (50.0–59.0). The median age of patients with chordoma was 54.0 (47.0–63.0) years. The crude incidence rate of mobile spine osteosarcoma was 0.04 per million. Conclusion: Ewing sarcoma was the most frequent primary osseous sarcoma of the mobile spine. A male predilection was observed among all major sarcomas of the mobile spine. Ewing sarcoma in Iran affects the mobile spine in slightly older ages compared to other studies. Myxoid chondrosarcoma is the most frequent subtype of the mobile spine chondrosarcoma. Chordoma affects male in older ages compared to females. In contrast with other studies which showed a bimodal distribution of osteosarcoma of the spine including young adult and older age groups, 86% of cases in Iran were in the age group of 10–40 years.

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16

Sbaraglia, Marta, Alberto Righi, Marco Gambarotti, and Angelo P. Dei Tos. "Ewing sarcoma and Ewing-like tumors." Virchows Archiv 476, no. 1 (December 4, 2019): 109–19. http://dx.doi.org/10.1007/s00428-019-02720-8.

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17

Dirksen, Uta, and Heribert Jürgens. "Approaching Ewing sarcoma." Future Oncology 6, no. 7 (July 2010): 1155–62. http://dx.doi.org/10.2217/fon.10.76.

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18

Jürgens, Heribert, and Uta Dirksen. "Ewing sarcoma treatment." European Journal of Cancer 47 (September 2011): S366—S367. http://dx.doi.org/10.1016/s0959-8049(11)70206-4.

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19

Pérez-González, Yosmar, Elena García-Esparza, Esther Conde, and Daniel Azorín. "Epiphyseal Ewing Sarcoma." International Journal of Surgical Pathology 21, no. 2 (July 25, 2012): 173–76. http://dx.doi.org/10.1177/1066896912453202.

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20

Shapeero, L. G., D. Vanel, M. Sundaram, L. V. Ackerman, P. Wuisman, T. W. Bauer, S. Neuenschwander, G. Contesso, C. Janney, and D. J. McDonald. "Periosteal Ewing sarcoma." Radiology 191, no. 3 (June 1994): 825–31. http://dx.doi.org/10.1148/radiology.191.3.8184073.

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21

Mascard, É., and J. M. Guinebretière. "Sarcoma de Ewing." EMC - Aparato Locomotor 34, no. 4 (January 2001): 1–14. http://dx.doi.org/10.1016/s1286-935x(01)72247-2.

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22

Mascard, E., N. Gaspar, and J. M. Guinebretière. "Sarcoma de Ewing." EMC - Aparato Locomotor 46, no. 1 (February 2013): 1–14. http://dx.doi.org/10.1016/s1286-935x(13)64171-4.

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23

Hoffmann, Christiane, Susanne Ahrens, J�rgen Dunst, Axel Hillmann, Winfried Winkelmann, Alan Craft, Ulrich G�bel, et al. "Pelvic Ewing sarcoma." Cancer 85, no. 4 (February 15, 1999): 869–77. http://dx.doi.org/10.1002/(sici)1097-0142(19990215)85:4<869::aid-cncr14>3.0.co;2-8.

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24

Mazur, Melissa A., Sridharan Gururangan, Julia A. Bridge, Thomas J. Cummings, Srinivasan Mukundan, Herbert Fuchs, Nicole Larrier, and Edward C. Halperin. "Intracranial Ewing sarcoma." Pediatric Blood & Cancer 45, no. 6 (2005): 850–56. http://dx.doi.org/10.1002/pbc.20430.

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25

Bonvin, Florent, and Laura Merlini. "PERIOSTEAL EWING SARCOMA." Pediatric Hematology and Oncology 24, no. 5 (January 2007): 369. http://dx.doi.org/10.1080/08880010701252915.

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26

O??CONNOR, MARY I., and DOUGLAS J. PRITCHARD. "Ewing??s Sarcoma." Clinical Orthopaedics and Related Research &NA;, no. 262 (January 1991): 78???87. http://dx.doi.org/10.1097/00003086-199101000-00011.

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27

Ahmed, Safia K., Steven I. Robinson, Scott H. Okuno, Peter S. Rose, and Nadia N. Issa Laack. "Adult Ewing Sarcoma." American Journal of Clinical Oncology 37, no. 5 (October 2014): 423–29. http://dx.doi.org/10.1097/coc.0b013e31827de65e.

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28

Labadie, Jessica G., Aleksandra G. Florek, Timothy VandenBoom, Pedram Yazdan, and Aleksandar L. Krunic. "Ewing-Like Sarcoma." American Journal of Dermatopathology 40, no. 12 (December 2018): 890–93. http://dx.doi.org/10.1097/dad.0000000000001237.

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29

Collier, Anderson B., Lesley Simpson, and Philip Monteleone. "Cutaneous Ewing Sarcoma." Journal of Pediatric Hematology/Oncology 33, no. 8 (December 2011): 631–34. http://dx.doi.org/10.1097/mph.0b013e31821b234d.

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30

Neilsen, Paul M., Kathleen I. Pishas, David F. Callen, and David M. Thomas. "Targeting the p53 Pathway in Ewing Sarcoma." Sarcoma 2011 (2011): 1–17. http://dx.doi.org/10.1155/2011/746939.

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The p53 tumour suppressor plays a pivotal role in the prevention of oncogenic transformation. Cancers frequently evade the potent antitumour surveillance mechanisms of p53 through mutation of theTP53gene, with approximately 50% of all human malignancies expressing dysfunctional, mutated p53 proteins. Interestingly, genetic lesions in theTP53gene are only observed in 10% of Ewing Sarcomas, with the majority of these sarcomas expressing a functional wild-type p53. In addition, the p53 downstream signaling pathways and DNA-damage cell cycle checkpoints remain functionally intact in these sarcomas. This paper summarizes recent insights into the functional capabilities and regulation of p53 in Ewing Sarcoma, with a particular focus on the cross-talk between p53 and the EWS-FLI1 gene rearrangement frequently associated with this disease. The development of several activators of p53 is discussed, with recent evidence demonstrating the potential of small molecule p53 activators as a promising systemic therapeutic approach for the treatment of Ewing Sarcomas with wild-type p53.

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31

Hack, Ruben I., Anton S. Becker, Beata Bode-Lesniewska, G. Ulrich Exner, Daniel A. Müller, Daniela A. Ferraro, Geoffrey I. Warnock, Irene A. Burger, and Christian Britschgi. "When SUV Matters: FDG PET/CT at Baseline Correlates with Survival in Soft Tissue and Ewing Sarcoma." Life 11, no. 9 (August 24, 2021): 869. http://dx.doi.org/10.3390/life11090869.

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Introduction: The role of positron-emission tomography/computed-tomography (PET/CT) in the management of sarcomas and as a prognostic tool has been studied. However, it remains unclear which metric is the most useful. We aimed to investigate if volume-based PET metrics (Tumor volume (TV) and total lesions glycolysis (TLG)) are superior to maximal standardized uptake value (SUVmax) and other metrics in predicting survival of patients with soft tissue and bone sarcomas. Materials and Methods: In this retrospective cohort study, we screened over 52′000 PET/CT scans to identify patients diagnosed with either soft tissue, bone or Ewing sarcoma and had a staging scan at our institution before initial therapy. We used a Wilcoxon signed-rank to assess which PET/CT metric was associated with survival in different patient subgroups. Receiver-Operating-Characteristic curve analysis was used to calculate cutoff values. Results: We identified a total of 88 patients with soft tissue (51), bone (26) or Ewing (11) sarcoma. Median age at presentation was 40 years (Range: 9–86 years). High SUVmax was most significantly associated with short survival (defined as <24 months) in soft tissue sarcoma (with a median and range of SUVmax 12.5 (8.8–16.0) in short (n = 18) and 5.5 (3.3–7.2) in long survival (≥24 months) (n = 31), with (p = 0.001). Similar results were seen in Ewing sarcoma (with a median and range of SUVmax 12.1 (7.6–14.7) in short (n = 6) and 3.7 (3.5–5.5) in long survival (n = 5), with (p = 0.017). However, no PET-specific metric but tumor-volume was significantly associated (p = 0.035) with survival in primary bone sarcomas (with a median and range of 217 cm3 (186–349) in short survival (n = 4) and 60 cm3 (22–104) in long survival (n = 19), with (p = 0.035). TLG was significantly inversely associated with long survival only in Ewing sarcoma (p = 0.03). Discussion: Our analysis shows that the outcome of soft tissue, bone and Ewing sarcomas is associated with different PET/CT metrics. We could not confirm the previously suggested superiority of volume-based metrics in soft tissue sarcomas, for which we found SUVmax to remain the best prognostic factor. However, bone sarcomas should probably be evaluated with tumor volume rather than FDG PET activity.

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32

Nagano, Akihito, Masanori Tsugita, Yutaka Nishimoto, Haruhiko Akiyama, and Akira Kawai. "The ‘other’ bone sarcomas in Japan: a retrospective study of primary bone sarcomas other than osteosarcoma, Ewing sarcoma and chondrosarcoma, using data from the Bone Tumuor Registry in Japan." Japanese Journal of Clinical Oncology 51, no. 9 (June 9, 2021): 1430–36. http://dx.doi.org/10.1093/jjco/hyab090.

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Abstract Background To date, no standard treatments for primary bone sarcomas other than those for osteosarcoma, Ewing sarcoma and chondrosarcoma have been developed. Methods The clinical characteristics and prognostic factors of 330 patients with primary bone sarcomas other than osteosarcoma, Ewing sarcoma and chondrosarcoma, listed in a nationwide tumour registry (Bone and Soft Tissue Tumor Registry in Japan) were investigated. The effects of adjuvant chemotherapy were determined by comparing the outcomes of patients with non-metastatic bone tumours who received surgery plus chemotherapy with those of patients who underwent surgery alone. Results The most common diagnosis was undifferentiated high-grade pleomorphic sarcoma. Axial site tumours (47.8%) and distant metastases at presentation (24.5%) were frequent. The 5-year overall and progression-free survival rates were 44.9 and 39.9%, respectively. Prognostic factor analysis identified surgery as an independent predictor of overall survival, and distant metastases at presentation was significant and independent predictor of both overall and progression-free survival. No significant difference in outcome was observed between patients treated with surgery alone and those treated with surgery plus chemotherapy (P = 0.71). Conclusions Patients with bone sarcomas other than osteosarcoma, Ewing sarcoma and chondrosarcoma without metastasis at presentation have a relatively good prognosis with definitive surgery; however, the benefit of adjuvant chemotherapy is unclear.

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33

Cross, Nathan M., A. Luana Stanescu, Erin R. Rudzinski, Douglas S. Hawkins, and Marguerite T. Parisi. "Vaginal Ewing Sarcoma: An Uncommon Clinical Entity in Pediatric Patients." Journal of Clinical Imaging Science 7 (April 25, 2017): 17. http://dx.doi.org/10.4103/jcis.jcis_96_16.

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Ewing sarcoma, including classical Ewing sarcoma of the bone and primitive neuroectodermal tumors arising in bone or extraosseous primary sites, is a highly aggressive childhood neoplasm. We present two cases of Ewing sarcoma arising from the vagina in young girls. Previously reported cases in literature focused on their pathologic rather than radiographic features. We describe the spectrum of multimodality imaging appearances of Ewing sarcoma at this unusual primary site. Awareness of vaginal Ewing tumors may facilitate prompt diagnosis and lead to a different surgical approach than the more commonly encountered vaginal rhabdomyosarcoma.

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Liu, Yen-Lin, Shu-Mei Chen, Hsin-Lun Lee, Jia-Hui Huang, Shu-Huey Chen, Yu-Chien Kao, Hsi Chang, et al. "RARE-45. SARCOMAS INVOLVING THE CENTRAL NERVOUS SYSTEM AT INITIAL PRESENTATION IN CHILDREN AND YOUNG ADULTS: A CASE SERIES." Neuro-Oncology 22, Supplement_3 (December 1, 2020): iii452. http://dx.doi.org/10.1093/neuonc/noaa222.755.

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Abstract Sarcomas of bone, soft tissue, or neural origin may occasionally invade the central nervous system (CNS), causing diagnostic and therapeutic challenges. We aim to investigate the clinical features of sarcomas involving the CNS at initial presentation. During 2015/01–2019/12, nine consecutive patients (4 Males and 5 Females) younger than 30 years of age treated at a University Healthcare System in Northern Taiwan were included. The median age was 8.7 years (range, 2–24 years); diagnoses were Ewing Sarcoma with EWSR1 rearrangements (n=4), CIC-NUTM1 Sarcoma (n=1), Osteosarcoma (n=2), Malignant Peripheral Nerve Sheath Tumor (MPNST; n=1), and extramedullary myeloid sarcoma (n=1). The tumors originated from the skull (n=1), dura (n=1), vertebra (n=4), spinal canal (n=1), or extra-CNS sites (n=2). Four patients had metastases (1 Ewing sarcoma, 2 osteosarcoma, and 1 extramedullary myeloid sarcoma). The main symptom at diagnosis was facial/eye pain (n=2), back pain (n=3), arm weakness (n=1), or gait disturbance (n=3). Upfront neurosurgical decompression (n=7) or urgent radiotherapy (n=1) was performed in most patients. At a median follow-up duration of 20.1 months, the overall survival rate was 70%. All patients with Ewing sarcoma (n=4) and CIC-NUTM1 sarcoma (n=1) achieved Complete Response after surgery, interval-compressed chemotherapy, radiotherapy, and adjuvant chemotherapy. Patients with stage IV osteosarcoma (n=2) had Partial Response; the patients with MPNST and extraskeletal myeloid sarcoma died of Progressive Disease at 18 and 3 months after diagnosis, respectively. We conclude that timely decompression, early diagnosis, and histology-driven multimodality treatment are effective strategies in managing sarcomas involving the CNS.

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35

Berning, Philipp, Carolin Hennemann, Claudia Tulotta, Christiane Schaefer, Birgit Lechtape, Marc Hotfilder, Yassmine El Gourari, et al. "The Receptor Tyrosine Kinase RON and Its Isoforms as Therapeutic Targets in Ewing Sarcoma." Cancers 12, no. 4 (April 7, 2020): 904. http://dx.doi.org/10.3390/cancers12040904.

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The receptor tyrosine kinase (RTK) RON is linked to an aggressive metastatic phenotype of carcinomas. While gaining interest as a therapeutic target, RON remains unstudied in sarcomas. In Ewing sarcoma, we identified RON among RTKs conferring resistance to insulin-like growth factor-1 receptor (IGF1R) targeting. Therefore, we explored RON in pediatric sarcoma cell lines and an embryonic Tg(kdrl:mCherry) zebrafish model, using an shRNA-based approach. To examine RON–IGF1R crosstalk, we employed the clinical-grade monoclonal antibody IMC-RON8, alone and together with the IGF1R-antibody IMC-A12. RON silencing demonstrated functions in vitro and in vivo, particularly within micrometastatic cellular capacities. Signaling studies revealed a unidirectional IGF1-mediated cross-activation of RON. Yet, IMC-A12 failed to sensitize cells to IMC-RON8, suggesting additional mechanisms of RON activation. Here, RT-PCR revealed that childhood sarcomas express short-form RON, an isoform resistant to antibody-mediated targeting. Interestingly, in contrast to carcinomas, treatment with DNA methyltransferase inhibitor did not diminish but increased short-form RON expression. Thus, this first report supports a role for RON in the metastatic progression of Ewing sarcoma. While principal molecular functions appear transferrable between carcinomas, Ewing sarcoma and possibly more common sarcoma subtypes, RON highlights that specific regulations of cellular networks and isoforms require better understanding to successfully transfer targeting strategies.

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36

Rahn, Douglas A., William Read, and Ajay Sandhu. "Treatment of Lung Metastases from Ewing Sarcoma with Stereotactic Body Radiotherapy rather than Whole-Lung Irradiation." Journal of Postgraduate Medicine, Education and Research 46, no. 1 (2012): 34–36. http://dx.doi.org/10.5005/jp-journals-10028-1008.

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ABSTRACT Advances in knowledge Lung metastases from Ewing sarcoma have traditionally been treated with whole-lung irradiation (WLI) and chemotherapy. WLI may unnecessarily treat uninvolved normal lung tissue and increase the risk of toxicity. Stereotactic body radiotherapy (SBRT) has proven to be a safe and effective treatment for primary lung cancer and lung metastases, but there is no significant published description of SBRT in lung metastases from Ewing sarcoma. Implications for patient care The patient presented in this case report had long-term survival from lung metastases of Ewing sarcoma treated with chemotherapy and SBRT. SBRT may prove to be safer and more effective than whole lung irradiation as a treatment of lung metastases in Ewing sarcoma. Summary statement Whether SBRT of lung metastases of Ewing sarcoma can improve outcomes and decrease toxicity compared to whole-lung irradiation, surgical resection or palliative chemotherapy merits further study. How to cite this article Rahn DA, Read W, Sandhu A. Treatment of Lung Metastases from Ewing Sarcoma with Stereotactic Body Radiotherapy rather than Whole-Lung Irradiation. J Postgrad Med Edu Res 2012;46(1):34-36.

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Hockertz, T., W. Eberl, and M. Velickovic. "Sacral Osteoneogenesis after Complete Sacrectomy in a Patient with Ewing Sarcoma." Case Reports in Orthopedics 2017 (2017): 1–7. http://dx.doi.org/10.1155/2017/7824687.

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Ewing sarcomas are the second most common primary malignant bone tumors in childhood and adolescence which rapidly metastasize. Due to improvement of treatment options in recent years, the survival rate has significantly increased. Nevertheless, lethality is still high, and neurologic symptoms are frequent. To the best of our knowledge, this is the first reported case of a sacral osteoneogenesis after complete sacrectomy in a patient with Ewing sarcoma.

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Bulbul, Ajaz, John Paul Shen, Joanne Xiu, Pablo Tamayo, and Hatim Husain. "Genomic and Proteomic Alterations in Desmoplastic Small Round Blue-Cell Tumors." JCO Precision Oncology, no. 2 (November 2018): 1–9. http://dx.doi.org/10.1200/po.17.00170.

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Purpose Desmoplastic small round blue-cell tumors (DSRCTs) are sarcomas that contain the t(11;22) (p13;q12) translocation EWS-WT1 fusion protein. Because this is a rare tumor type, prospective clinical trials in DSRCT are challenging. Patients are treated in a manner similar to those with Ewing sarcoma; however, differences in prognosis and clinical presentation suggest fundamental differences in biology and potentially different therapeutic implications. This study aimed to characterize the molecular characteristics of DSRCT tumors to explore unique therapeutic options for this extremely rare and aggressive cancer type. Methods Thirty-five DSRCT tumors were assessed using next-generation sequencing, protein expression (immunohistochemistry), and gene amplification (chromogenic in situ hybridization or fluorescence in situ hybridization). Three patients had tumor mutational load, which was calculated as somatic nonsynonymous missense mutations sequenced with a 592-gene panel. Gene expression data were obtained for an additional seven DSRCT tumors. Molecular alterations were compared with 88 Ewing sarcomas. Results The most common alterations that distinguished DSRCTs from Ewing sarcoma included higher androgen receptor (AR), TUBB3, epidermal growth factor receptor, and TOPO2A expression. Independent analysis by RNA sequencing confirmed higher AR expression from an independent data set of EWS-WT1 fusion–positive DSRCTs compared with Ewing sarcoma and a pan-cancer analysis. DSRCTs had somatic mutations that were identified in TP53 and FOXO3, averaged five mutations per megabase, and no programmed death-ligand 1 expression was detected in any DSRCT samples. Conclusion The current analysis provides the first comparative analysis, to our knowledge, of molecular aberrations that distinguish DSRCT from Ewing sarcoma. High AR expression seems to be a defining event in these malignancies, and additional investigation of the responsiveness of AR inhibitors in this disease is encouraged.

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Hesla, Asle Charles, Andri Papakonstantinou, and Panagiotis Tsagkozis. "Current Status of Management and Outcome for Patients with Ewing Sarcoma." Cancers 13, no. 6 (March 10, 2021): 1202. http://dx.doi.org/10.3390/cancers13061202.

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Ewing sarcoma is the second most common bone sarcoma in children after osteosarcoma. It is a very aggressive malignancy for which systemic treatment has greatly improved outcome for patients with localized disease, who now see survival rates of over 70%. However, for the quarter of patients presenting with metastatic disease, survival is still dismal with less than 30% of patients surviving past 5 years. Patients with disease relapse, local or distant, face an even poorer prognosis with an event-free 5-year survival rate of only 10%. Unfortunately, Ewing sarcoma patients have not yet seen the benefit of recent years’ technical achievements such as next-generation sequencing, which have enabled researchers to study biological systems at a level never seen before. In spite of large multinational studies, treatment of Ewing sarcoma relies entirely on chemotherapeutic agents that have been largely unchanged for decades. As many promising modern therapies, including monoclonal antibodies, small molecules, and immunotherapy, have been disappointing to date, there is no clear candidate as to which drug should be investigated in the next large-scale clinical trial. However, the mechanisms driving tumor development in Ewing sarcoma are slowly unfolding. New entities of Ewing-like tumors, with fusion transcripts that are related to the oncogenic EWSR1-FLI1 fusion seen in the majority of Ewing tumors, are being mapped. These tumors, although sharing much of the same morphologic features as classic Ewing sarcoma, behave differently and may require a different treatment. There are also controversies regarding local treatment of Ewing sarcoma. The radiosensitive nature of the disease and the tendency for Ewing sarcoma to arise in the axial skeleton make local treatment very challenging. Surgical treatment and radiotherapy have their pros and cons, which may give rise to different treatment strategies in different centers around the world. This review article discusses some of these controversies and reproduces the highlights from recent publications with regard to diagnostics, systemic treatment, and surgical treatment of Ewing sarcoma.

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Pishas, Kathleen I., and Stephen L. Lessnick. "Recent advances in targeted therapy for Ewing sarcoma." F1000Research 5 (August 25, 2016): 2077. http://dx.doi.org/10.12688/f1000research.8631.1.

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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.

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Rama-López, Julio, Rafael Ramos Asensio, Cesar García-Garza, Pablo Luna Fra, Maria del Carmen Gassent Balaguer, and Jos& Fuster Salva. "Extraosseous Ewing Sarcoma: Expanding the Differential Diagnosis of Supraclavicular Fossa Tumors." Ear, Nose & Throat Journal 96, no. 1 (January 2017): E29—E32. http://dx.doi.org/10.1177/014556131709600104.

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A broad spectrum of diseases can be included in the differential diagnosis of neck masses. We report a case of extraosseous Ewing sarcoma that presented as a neck mass in a 70-year-old man. To the best of our knowledge, this is the first reported case of extraosseous Ewing sarcoma of the supraclavicular fossa. Published cases of extraosseous Ewing sarcoma in the neck have been described in other age groups, but those tumors were confined to the parapharyngeal space. Also, there have been reported cases in patients older than 70 years in which Ewing sarcoma affected other structures such as the larynx and the pelvis, but none in the soft tissues of the neck. This case adds extraosseous Ewing sarcoma as a possible diagnosis to consider when evaluating a neck mass in the supraclavicular fossa.

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Petrovic, Marija, Bihong Zhao, Manoj Thangam, Pranav Loyalka, L. Maximilian Buja, Biswajit Kar, and Igor D. Gregoric. "Ewing Sarcoma in the Right Ventricle." Texas Heart Institute Journal 43, no. 5 (October 1, 2016): 458–60. http://dx.doi.org/10.14503/thij-15-5330.

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Ewing sarcoma is the second most prevalent malignant primary bone tumor but constitutes only a small proportion of cardiac metastases. We present a case of asymptomatic Ewing sarcoma metastatic to the right ventricle. A 36-year-old man presented for evaluation and resection of a pedunculated right ventricular cardiac tumor. Three years before, he had been diagnosed with translocation-negative Ewing sarcoma, for which he had undergone chemotherapy and amputation of the left leg below the knee. We resected the right ventricular tumor. Analysis of the resected mass supported the diagnosis of metastatic Ewing sarcoma. Postoperative transthoracic echocardiograms showed normal biventricular size and function. One year later, the patient had no recurrence of the sarcoma. In addition to discussing this case, we review the relevant medical literature.

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Klimo, Paul, Patrick J. Codd, Holcombe Grier, and Liliana C. Goumnerova. "Primary pediatric intraspinal sarcomas." Journal of Neurosurgery: Pediatrics 4, no. 3 (September 2009): 222–29. http://dx.doi.org/10.3171/2009.3.peds08272.

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Sarcomas that arise from within the spinal canal are rare, particularly within the pediatric population. In general, these primary intraspinal sarcomas are highly aggressive, posing unique treatment challenges with respect to surgery and choice of adjuvant therapy. The goal must be to obtain the most complete resection possible to minimize the risk of recurrence and metastasis, while preventing potential neurological deficits that may result from aggressive surgery. Among these primary intraspinal sarcomas are malignant peripheral nerve sheath tumors and members of the Ewing sarcoma family of tumors. The authors present 3 cases of unique spinal sarcomas in children—2 malignant peripheral nerve sheath tumors in patients without neurofibromatosis and an intradural extraosseous Ewing sarcoma arising from the sensory component of a lumbar spinal nerve—and discuss their management and outcome with a review of the current literature.

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Kim, Hyung Woo, Hong Ju Sohn, Mee Joo, Yun Kyung Kang, Woo Ki Jeon, Hyuk Pyo Lee, Joo In Kim, Soo Jeon Choi, and Ho Kee Yum. "Extraskeletal Ewing`s sarcoma Extraskeletal Ewings Sarcoma Arising in the Chest Wall." Tuberculosis and Respiratory Diseases 44, no. 5 (1997): 1158. http://dx.doi.org/10.4046/trd.1997.44.5.1158.

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Karikari, Isaac O., Ankit I. Mehta, Shahid Nimjee, Tiffany R. Hodges, June Tibaleka, Charita Montgomery, Lauren Simpson, Thomas J. Cummings, and Carlos A. Bagley. "Primary Intradural Extraosseous Ewing Sarcoma of the Spine: Case Report and Literature Review." Neurosurgery 69, no. 4 (May 12, 2011): E995—E999. http://dx.doi.org/10.1227/neu.0b013e318223b7c7.

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Abstract BACKGROUND AND IMPORTANCE: To report a rare case of spinal intradural extraosseous Ewing sarcoma in an adult and review current literature. Although Ewing sarcoma belongs to the family, the treatment modalities are different, and thus the correct diagnosis is very important despite its rare occurrence. CLINICAL PRESENTATION: A 56-year-old woman presented with nocturnal bilateral buttock and leg pain. Magnetic resonance imaging (MRI) showed an enhancing intradural extramedullary extraosseous tumor at L1. INTERVENTION: A T12-L2 laminectomy was performed to resect the tumor. Immunohistochemical analysis confirmed the diagnosis of Ewing sarcoma. A thorough diagnostic workup did not reveal any bony origin of the tumor. Primary intradural central nervous system Ewing sarcoma is infrequently encountered and shares imaging and histopathological features with central primitive neuroectodermal tumors. Establishment of the right diagnosis is crucial because it mandates a distinct workup and treatment modality different from that for central primitive neuroectodermal tumor. Although osseous Ewing sarcoma predominantly occurs in children and young adults, extraosseous central nervous system Ewing sarcoma is not uncommon in adults and should therefore be considered in the differential diagnosis of extraosseous small blue cell tumors in adult patients.

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Fernández del Castillo Ascanio, M., and S. Sirvent Cerdá. "Sarcoma de Ewing extraóseo." Radiología 52, no. 3 (May 2010): 276–77. http://dx.doi.org/10.1016/j.rx.2010.02.007.

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Bancalari, Eugenia, Enrique de Álava, and Juan C. Tardío. "Primary Vaginal Ewing Sarcoma." International Journal of Surgical Pathology 20, no. 3 (October 17, 2011): 305–10. http://dx.doi.org/10.1177/1066896911424898.

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Aymor??, Ierec?? Lins, Walter Meohas, Ana Luzia Brito de Almeida, and Danielle Proebstner. "Periosteal Ewing???s Sarcoma." Clinical Orthopaedics and Related Research &NA;, no. 434 (May 2005): 265–72. http://dx.doi.org/10.1097/01.blo.0000151439.18746.f1.

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Sánchez-Legaza, Elena, Rosario Guerrero-Cauqui, and Herminia Revelles-Suarez. "Sarcoma de Ewing hipofaríngeo." Acta Otorrinolaringológica Española 71, no. 5 (September 2020): 324–25. http://dx.doi.org/10.1016/j.otorri.2019.08.004.

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Sciubba, Daniel M., Scott H. Okuno, Mark B. Dekutoski, and Ziya L. Gokaslan. "Ewing and Osteogenic Sarcoma." Spine 34, Supplement (October 2009): S58—S68. http://dx.doi.org/10.1097/brs.0b013e3181ba6436.

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