Primary Renal Ewing Sarcoma in Children and Young Adults

Bradford, Kathryn; Nobori, Alexander; Johnson, Brittany; Allen‐Rhoades, Wendy; Naik‐Mathuria, Bindi; Panosyan, Eduard H.; Gotesman, Moran; Lasky, Joseph L.; Cheng, Jerry C.; Ikeda, Alan K.; Goldstein, Jeffrey; Singh, Arun S.; Federman, Noah

doi:10.1097/mph.0000000000001804

Cited by 11 publications

(13 citation statements)

References 25 publications

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“…The Ewing sarcoma family of tumors (ESFT) is a collection of small, rounded tumor cells that have similar neural histological and genetic characteristics [ 1 , 2 , 3 , 4 ]. ESFT is categorized into four types based on the origin of the tumor: Ewing sarcoma of the bone, peripheral primitive neuroectodermal tumor (pPNET), Askin tumor, which originates from the chest wall, and, finally, the extraosseous or extraskeletal Ewing sarcoma (EES).…”

Section: Introductionmentioning

confidence: 99%

Extraosseous Ewing Sarcoma in Children: A Systematic Review and Meta-Analysis of Clinicodemographic Characteristics

Ghandour

Lehner

Klotz³

et al. 2022

Children

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Background: We conducted this systematic review to provide comprehensive evidence on the prevalence, clinical features and outcomes of young extraosseous Ewing sarcoma (EES) cases. Methods: PubMed, Scopus, Web of Science, and Google Scholar were searched for articles reporting the occurrence of EES among children and adolescents (<21 years). The primary outcome included the rate of occurrence of EES among children and adolescents, while the secondary outcomes included the descriptive analyses of the demographic characteristics, tumor characteristics, and clinical outcomes of the affected cases. The data are reported as the effect size (ES) and its corresponding 95% confidence interval (CI). Results: A total of 29 studies were included. Twenty-four reported instances of childhood disease among all the EES cases [ES = 30%; 95%CI: 29–31%], while five studies reported extraosseous cases among the pediatric EES cases [ES = 22%; 95%CI: 13–31%]. The thorax is the most common location of childhood EES [33%; 95%CI: 20–46%] followed by the extremities [31%; 95%CI: 22–40%]. Concurrent chemotherapy and radiotherapy [57%; 95% CI: 25–84%] was the most commonly implemented management protocol in the pediatric EES cases. The rate of no evidence of disease and 5-year overall survival was 69% for both outcomes. Mortality occurred in 29% of cases, while recurrence and secondary metastasis occurred in 35% and 16% of cases, respectively. Conclusions: Our findings provide insight into the clinical features and outcomes of EES among children and adolescents.

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Section: Introductionmentioning

confidence: 99%

Extraosseous Ewing Sarcoma in Children: A Systematic Review and Meta-Analysis of Clinicodemographic Characteristics

Ghandour

Lehner

Klotz³

et al. 2022

Children

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“…Ewing sarcoma ranks second in the incidence of primary malignant bone tumors among children and adolescents, and its proportion in primary bone tumors is 6% to 8% [1]. After traditional surgery, radiotherapy and chemotherapy, most patients will die within 2 years, and the five-year survival rate is under 10% [2,3]. After IGF-1R is activated and overexpressed in Ewing sarcoma, it may have a synergistic effect on the fusion gene EWS-FLI1, thus driving the occurrence of Ewing sarcoma [4,6].…”

Section: Discussionmentioning

confidence: 99%

“…The current primary treatments for Ewing sarcoma are surgery alone, radiotherapy, and single-agent chemotherapy [2]. However, none of the treatments have favorable outcomes, and most patients will die within 2 years, with 5-year survival rates of no more than 10% [3]. Therefore, there is a need to develop more effective treatment strategies for Ewing sarcoma.…”

Section: Introductionmentioning

confidence: 99%

Effective natural inhibitors targeting IGF-1R by computational study

Wang

Zhou

Lin

et al. 2022

Aging

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IGF-1R belongs to a tyrosine kinase family and is currently a newly discovered drug target. IGF-1R inhibitors can bind directly to IGF-1R to achieve the effect of inhibiting the function of IGF-1R. At present, IGF-1R inhibitors have good clinical effects on Ewing sarcoma in the clinic. In this article, we screened compounds capable of inhibiting IGF-1R function through computer-aided virtual technology. First, some molecules with good docking properties for IGF-1R can be screened by LibDock. Then, ADME analysis (adsorption, distribution, metabolism, and excretion) and toxicity indicators were performed. The mechanism of binding and the binding affinity in the middle of IGF-1R and ligand were verified using molecular docking. Ultimately, the stability of ligand-receptor complex was evaluated using molecular dynamics simulations. In line with the results, two natural compounds ZINC000014946303 and ZINC000006003042 were found in the ZINC database, potential effective inhibitors of IGF-1R. ZINC000014946303 and ZINC000006003042 can bind to IGF-1R with high binding affinity as predicted by molecular docking. It was also found that they are not hepatotoxic, with less developmental toxicity potential, rodent carcinogenicity, Ames mutagenicity, and high tolerance to cytochrome P4502D6. Hereby, this study aimed to screen out ideal compounds that have inhibitory effects on IGF-1R from the drug library and, at the same time, provide a direction for the future development of IGF-1R inhibitors.

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“…Traditional therapy methods for Ewing sarcoma, such as surgery, radiotherapy, and chemotherapy, are hard to reach notably satisfactory results [4]. The ve-year survival rate for patients is less than 10 percent, and most patients are prone to dying within two years [5]. We should focus on nding interesting therapeutic strategies for Ewing sarcoma.…”

Section: Introductionmentioning

confidence: 99%

Effective natural inhibitors targeting LSD1 by computational study

Wang

Lin

Zhou

et al. 2023

Preprint

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Objective: This study aimed to screen lead compounds and drug candidates with an inhibitory effect on the function of LSD1 from the ZINC database. Methods: We used computer-aided virtual technology to screen some agents that inhibit the function of LSD1. Initially, LibDock screened out some optimal compounds for docking with LSD1. These candidate compounds were subjected to ADME analysis (adsorption, distribution, metabolism, and excretion) and toxicity metrics. Molecular docking can determine the binding affinity between LSD1 and the ligand, and lastly, we applied molecular dynamics simulations to calculate the docking of the ligand-receptor complex. Results: Two natural compounds, ZINC000001651126 and ZINC000000001083, found in the ZINC database, are potent inhibitors of LSD1. When ZINC000001651126 and ZINC000000001083 bind to LSD1, they show high binding affinity. They are not hepatotoxic and have a high tolerance to cytochrome P4502D6. In addition, ZINC000001651126 and ZINC000000001083 have less developmental toxicity potential, rodent carcinogenicity, and Ames mutagenicity. Conclusions: ZINC000001651126 and ZINC000000001083 can be considered safe and ideal drug candidates for LSD1 inhibitors. This study can provide new ideas for future research and the application of LSD1 inhibitors.

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Primary Renal Ewing Sarcoma in Children and Young Adults

Cited by 11 publications

References 25 publications

Extraosseous Ewing Sarcoma in Children: A Systematic Review and Meta-Analysis of Clinicodemographic Characteristics

Extraosseous Ewing Sarcoma in Children: A Systematic Review and Meta-Analysis of Clinicodemographic Characteristics

Effective natural inhibitors targeting IGF-1R by computational study

Effective natural inhibitors targeting LSD1 by computational study

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