4.7 Article

Outcome for adolescent and young adult patients with osteosarcoma

Journal

CANCER
Volume 118, Issue 18, Pages 4597-4605

Publisher

WILEY-BLACKWELL
DOI: 10.1002/cncr.27414

Keywords

osteosarcoma; adolescent; young adult; prognosis; outcome

Categories

Funding

  1. Children's Oncology Group Chair's grant [NIH U10 CA98543]
  2. Human Specimen Banking grant [NIH U24 CA114766]
  3. WWWW (QuadW) Foundation
  4. Aflac/CureSearch for Children's Cancer AYA Cancer Research Program
  5. Timothy O'Brien Trust Award
  6. St. Baldrick's Foundation

Ask authors/readers for more resources

BACKGROUND: There are conflicting data regarding age as a prognostic factor in osteosarcoma. The authors conducted a study evaluating the impact of age on prognosis in children and young adults with osteosarcoma enrolled on North American cooperative group trials. METHODS: Patients with high-grade osteosarcoma of any site enrolled on North American cooperative group trials CCG-7943, POG-9754, INT-0133, and AOST0121 were included in this study. Primary tumor site, age, sex, ethnicity, histologic response, and presence of metastatic disease at diagnosis were evaluated for their impact on overall survival (OS) and event-free survival (EFS). RESULTS: A total of 1054 patients were eligible and had complete data available for the study. Age was not significantly associated with any other presenting covariate analyzed except sex. Age 18 or older was associated with a statistically significant poorer EFS (P = .019) and OS (P = .043). The 10-year EFS and OS in patients <10, 10 to 17, and =18 years old were 55%, 55%, 37% and 68%, 60%, 41%, respectively. The poorer EFS in patients =18 years old was because of an increased rate of relapse. Presence of metastatic disease at diagnosis, poor histologic response, and pelvic tumor site were also associated with a poorer prognosis. In multivariate analysis, age continued to be associated with poorer EFS (P = .019) and OS (P = .049). CONCLUSIONS: In osteosarcoma, age 18 to 30 years is associated with a statistically significant poorer outcome because of an increased rate of relapse. Poorer outcome in adolescent and young adult patients is not explained by tumor location, histologic response, or metastatic disease at presentation. Cancer 2012.(c) 2012 American Cancer Society.

Authors

I am an author on this paper
Click your name to claim this paper and add it to your profile.

Reviews

Primary Rating

4.7
Not enough ratings

Secondary Ratings

Novelty
-
Significance
-
Scientific rigor
-
Rate this paper

Recommended

Article Pathology

Deep Learning of Rhabdomyosarcoma Pathology Images for Classification and Survival Outcome Prediction

Xinyi Zhang, Shidan Wang, Erin R. Rudzinski, Saloni Agarwal, Ruichen Rong, Donald A. Barkauskas, Ovidiu Daescu, Lauren Furman Cline, Rajkumar Venkatramani, Yang Xie, Guanghua Xiao, Patrick Leavey

Summary: This study developed a computational algorithm for subtype classification and prognosis prediction of rhabdomyosarcoma (RMS) using pathology images. The algorithm achieved good performance in classification and prediction on the test dataset, providing assistance in pathology evaluation and risk stratification.

AMERICAN JOURNAL OF PATHOLOGY (2022)

Letter Oncology

Reply to R. Lakhotia et al

G. A. Amos Burke, Veronique Minard-Colin, Anne Auperin, Sarah Alexander, Marta Pillon, Rafael Delgado, Jozsef Zsiros, Anne Uyttebroeck, Peggy Dartigues, Rodney R. Miles, Bernarda Kazanowska, Alan K. Chiang, Stephanie Haouy, Catherine M. Bollard, Monika Csoka, Keith Wheatley, Donald A. Barkauskas, Peter C. Adamson, Gilles Vassal, Catherine Patte, Thomas G. Gross

JOURNAL OF CLINICAL ONCOLOGY (2022)

Article Oncology

Membrane-Anchored and Tumor-Targeted IL12 (attIL12)-PBMC Therapy for Osteosarcoma

Qing Yang, Jiemiao Hu, Zhiliang Jia, Qi Wang, Jing Wang, Long Hoang Dao, Wendong Zhang, Sheng Zhang, Xueqing Xia, Richard Gorlick, Shulin Li

Summary: This study proposes a new cell therapy for treating osteosarcoma. By introducing a specific protein into peripheral blood mononuclear cells, tumor growth can be effectively inhibited without observable toxicity.

CLINICAL CANCER RESEARCH (2022)

Article Biochemistry & Molecular Biology

Integrated DNA Copy Number and Expression Profiling Identifies IGF1R as a Prognostic Biomarker in Pediatric Osteosarcoma

Aaron M. Taylor, Jiayi M. Sun, Alexander Yu, Horatiu Voicu, Jianhe Shen, Donald A. Barkauskas, Timothy J. Triche, Julie M. Gastier-Foster, Tsz-Kwong Man, Ching C. Lau

Summary: This study revealed the heterogeneity of osteosarcoma in younger populations and identified several genes, such as IGF1R and FGFR2, that are associated with the pathogenesis of the disease. These genes were found to have a significant impact on the prognosis of patients.

INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES (2022)

Article Oncology

Adverse prognostic impact of the loss of STAG2 protein expression in patients with newly diagnosed localised Ewing sarcoma: A report from the Children's Oncology Group

David S. Shulman, Sonja Chen, David Hall, Anwesha Nag, Aaron R. Thorner, Stephen L. Lessnick, Kimberly Stegmaier, Katherine A. Janeway, Steven G. DuBois, Mark D. Krailo, Donald A. Barkauskas, Alanna J. Church, Brian D. Crompton

Summary: This study aimed to determine the frequency of STAG2 protein loss and its association with the outcome of Ewing sarcoma. The results showed that STAG2 expression loss is common and associated with a poor prognosis in patients without STAG2 mutations. In addition, STAG2 loss in local disease is associated with a decrease in the 5-year event-free survival rate.

BRITISH JOURNAL OF CANCER (2022)

Article Oncology

Irinotecan dose schedule for the treatment of Ewing sarcoma

Paul Meyers, Emily Slotkin

Summary: This study reviewed the use of irinotecan and temozolomide in patients with recurrent Ewing sarcoma. The results showed that a 10-day irinotecan treatment schedule had a higher objective response rate compared to a 5-day schedule. Therefore, the use of a 10-day irinotecan schedule should be considered in the treatment of recurrent Ewing sarcoma.

PEDIATRIC BLOOD & CANCER (2023)

Article Pathology

Deep Learning-Based Objective and Reproducible Osteosarcoma Chemotherapy Response Assessment and Outcome Prediction

David J. Ho, Narasimhan P. Agaram, Stephanie D. Suser, Cynthia Chu, Chad M. Vanderbilt, Paul A. Meyers, Leonard H. Wexler, John H. Healey, Thomas J. Fuchs, Meera R. Hameed

Summary: In this study, a deep learning-based approach was proposed to estimate necrosis ratio and predict treatment response and patient outcome in osteosarcoma. The results demonstrate that deep learning can serve as an objective tool for pathologists to analyze histology, assess treatment response, and predict patient prognosis.

AMERICAN JOURNAL OF PATHOLOGY (2023)

Article Oncology

Predicting Molecular Subtype and Survival of Rhabdomyosarcoma Patients Using Deep Learning of H&E Images: A Report from the Children's Oncology Group

David Milewski, Hyun Jung, G. Thomas Brown, Yanling Liu, Ben Somerville, Curtis Lisle, Marc Ladanyi, Erin R. Rudzinski, Hyoyoung Choo-Wosoba, Donald A. Barkauskas, Tammy Lo, David Hall, Corinne M. Linardic, Jun S. Wei, Hsien-Chao Chou, Stephen X. Skapek, Rajkumar Venkatramani, Peter K. Bode, Seth M. Steinberg, George Zaki, Igor B. Kuznetsov, Douglas S. Hawkins, Jack F. Shern, Jack Collins, Javed Khan

Summary: This study utilized convolutional neural networks (CNN) to predict high-risk mutations and prognosis of rhabdomyosarcoma (RMS) based on histologic features learned from H&E images. The CNN model achieved superior performance in predicting survival and event-free outcomes compared to current molecular-clinical risk stratification methods.

CLINICAL CANCER RESEARCH (2023)

Article Oncology

Lymph node metastases in paediatric and young adult patients with non-rhabdomyosarcoma soft tissue sarcoma (NRSTS): Findings from Children?s Oncology Group (COG) study ARST0332

Elysia Alvarez, Jiayi He, Sheri L. Spunt, Andrea Hayes-Jordan, Simon C. Kao, David M. Parham, Lynn Million, Aaron R. Weiss, Donald A. Barkauskas

Summary: The aim of this study is to define the clinical features and outcomes of young patients with NRSTS with LN metastases. LN metastases occur in about 4% of paediatric/young adult NRSTS patients, are limited to a few histologic subtypes, and are rare in patients without clinical or imaging evidence of lymphadenopathy. Therefore, biopsies of non-enlarged LNs are not necessary to identify occult involvement.

EUROPEAN JOURNAL OF CANCER (2023)

Correction Medicine, General & Internal

Osteosarcoma (vol 8, 77, 2022)

Hannah C. Beird, Stefan S. Bielack, Adrienne M. Flanagan, Jonathan Gill, Dominique Heymann, Katherine A. Janeway, J. Andrew Livingston, Ryan D. Roberts, Sandra J. Strauss, Richard Gorlick

NATURE REVIEWS DISEASE PRIMERS (2022)

Article Medicine, General & Internal

Osteosarcoma A

Hannah C. Beird, Stefan S. Bielack, Adrianne M. Flanagan, Jonathan Gill, Dominique Heymann, Katherine A. Janeway, J. Andrew Livingston, Ryan D. Roberts, Sandra J. Strauss, Richard Gorlick

Summary: Osteosarcoma is the most common primary malignant bone tumor with a bimodal incidence peaking at 18 and 60 years of age. It is more common in males and is driven by genetic factors related to bone formation leading to malignant progression and metastasis. Screening is currently focused on high-risk groups, and the prognosis for patients with metastatic disease remains poor.

NATURE REVIEWS DISEASE PRIMERS (2022)

Article Biochemistry & Molecular Biology

The Role of FAS Receptor Methylation in Osteosarcoma Metastasis

Jiayi Sun, Wing-Yuk Chow, Gufeng Xu, M. John Hicks, Manjula Nakka, Jianhe Shen, Patrick Kwok Shing M. Ng, Aaron Taylor, Alexander E. Yu, Jason A. Farrar, Donald Barkauskas, Richard M. Gorlick, Jaime M. Guidry Auvil, Daniela Gerhard, Paul Meltzer, Rudy Guerra, Tsz-Kwong C. Man, Ching Lau, TARGET Osteosarcoma Consortium

Summary: Osteosarcoma is a primary malignant bone tumor that commonly metastasizes to the lungs. Altered FAS expression due to DNA methylation has been explored as a mechanism in cancer cells. Analysis revealed high variability in methylation of CpG sites that correlated with FAS mRNA expression. Treatment with demethylating agent 5-azacytidine restored FAS protein expression and reduced metastasis in osteosarcoma cells.

INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES (2023)

Article Oncology

Prospective Clinical Genomic Profiling of Ewing Sarcoma: ERF and FGFR1 Mutations as Recurrent Secondary Alterations of Potential Biologic and Therapeutic Relevance

Koichi Ogura, Arielle Elkrief, Anita S. Bowman, Richard P. Koche, Elisa de Stanchina, Ryma Benayed, Audrey Mauguen, Marissa S. Mattar, Inna Khodos, Paul A. Meyers, John H. Healey, William D. Tap, Meera Hameed, Ahmet Zehir, Neerav Shukla, Charles Sawyers, Rohit Bose, Emily Slotkin, Marc Ladanyi

Summary: This study analyzed the cooperating secondary genetic alterations in Ewing sarcoma (ES), identifying recurrent alterations in ERF and FGFR1 genes. Functional studies showed that ERF alterations influenced tumor cell growth, colony formation, and motility, while loss of ERF promoted cellular proliferation and clonogenic growth. The findings provide new insights into ES pathobiology and potential therapeutic approaches for a subset of ES patients.

JCO PRECISION ONCOLOGY (2022)

Meeting Abstract Oncology

Identification of common germline variants associated with pediatric rhabdomyosarcoma survival: A report from the Children's Oncology Group (COG)

Bailey A. Martin-Giacalone, Michael E. Scheurer, Javed Khan, Stephen J. Chanock, Shengchao Alfred Li, Meredith Yeager, Deborah A. Marquez-Do, Donald A. Barkauskas, David Hall, Matthew T. McEvoy, Melissa A. Richard, Pagna Sok, Austin L. Brown, Aniko Sabo, Stephen X. Skapek, Douglas S. Hawkins, Rajkumar Venkatramani, Lisa Mirabello, Philip J. Lupo

CANCER RESEARCH (2022)

No Data Available