Descriptive epidemiology and outcomes of bone sarcomas in adolescent and young adult patients in Japan

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Abstract

Background

There have been fewer improvements in the clinical outcomes of adolescent and young adult (AYA) patients with cancer than for children and older adults, possibly because fewer studies focus on patients in this age group. The aims of this study were (1) to determine survival rates of bone sarcoma among AYAs in Japan (for comparison with other age groups), and (2) to establish whether belonging to the AYA age group at diagnosis was correlated with poor cancer survival in Japan.

Methods

A total of 3457 patients diagnosed with bone sarcoma (1930 male and 1527 female) were identified from 63,931 records in the Bone and Soft Tissue Tumor (BSTT) registry, a nationwide Japanese database, from 2006 to 2013. The histologic subtypes of bone sarcoma were osteosarcoma, chondrosarcoma, and Ewing sarcoma. The primary endpoints for prognosis were the occurrence of tumor-related death. We compared the epidemiological features of AYAs with other age groups. The cancer survival rates were calculated using the Kaplan-Meier method. Cox proportional hazards models were used to analyze the prognostic factors for cancer survival.

Results

The majority of AYA had osteosarcoma 631 (56.2%), while 198 (17.6%) had chondrosarcoma. The frequency of bone sarcoma occurrence was highest among AYA patients, who accounted for a marked proportion of patients with each type of sarcoma. With the exception of sarcoma type, AYA patients did not significantly differ from patients in other age groups for any of the investigated clinicopathological parameters. Cancer survival of AYA patients was significantly higher than in the elderly. Univariate and multivariate analyses revealed that AYA status was not a predictor of poor cancer survival. However, older age (≥65 years) was a predictor of poor cancer survival in patients with overall bone sarcoma, osteosarcoma, chondrosarcoma.

Conclusion

This epidemiological study is the first to investigate AYA patients with bone sarcoma using the nationwide BSTT Registry. We found that cancer survival of AYA patients was significantly higher than that of the elderly. AYA status was not a predictor of poor cancer survival in Japan.

Electronic supplementary material

The online version of this article (10.1186/s12891-018-2217-1) contains supplementary material, which is available to authorized users.

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Most cited references 21

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Descriptive epidemiology of sarcomas in Europe: Report from the RARECARE project

C.A. Stiller, A. Trama, D. Serraino … (2013)

Sarcomas are a heterogeneous group of malignant neoplasms arising from mesenchymal cells which encompass dozens of histological types and can occur in virtually any anatomic site. They form one of the principal groups of rare cancers in Europe as defined in the RARECARE project. We analysed 45,568 incident cases diagnosed during 1995-2002 and registered by 76 population-based cancer registries. Total crude incidence was 5.6 per 100,000 per year with an estimated 27,908 new cases per year in the EU27 countries, of which 84% were soft tissue sarcomas and 14% were bone sarcomas. Gastrointestinal stromal tumours (GIST) were only widely recognised as an entity in the late 1990s and consequently were under-registered. Their true incidence is believed to be about 1.5 per 100,000. Age-standardised incidence of soft tissue sarcomas ranged from 3.3 per 100,000 in Eastern Europe to 4.7 per 100,000 in Northern Europe. About 280,000 persons were estimated to be alive at the beginning of 2003 with a past diagnosis of sarcoma, of which 83% were soft tissue sarcomas and 16% were bone sarcomas. Five-year relative survival for 2000-2002 by the period was 58% for soft tissue sarcomas and 62% for bone sarcomas. The diversity and rarity of sarcomas combined with the quite large number of people affected by them mean that they provide a classic example of the importance of networking in diagnosis, therapy and research for rare cancers. Copyright © 2012 Elsevier Ltd. All rights reserved.

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The distinctive biology of cancer in adolescents and young adults.

Archie Bleyer, Ronald Barr, Brandon Hayes-Lattin … (2008)

One explanation for the relative lack of progress in treating cancer in adolescents and young adults is that the biology of malignant diseases in this age group is different than in younger and older persons, not only in the spectrum of cancers but also within individual cancer types and within the patient (host). Molecular, epidemiological and therapeutic outcome comparisons offer clues to this distinctiveness in most of the common cancers of adolescents and young adults. Translational and clinical research should not assume that the biology of cancers and patients is the same as in other age groups, and treatment strategies should be tailored to the differences.

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Chondrosarcoma in the United States (1973 to 2003): an analysis of 2890 cases from the SEER database.

A Giuffrida, Leonidas Koniaris, Robert Duncan … (2009)

Current demographic, prognostic, and outcomes data on the diagnosis and treatment of chondrosarcoma have been based on case series reported by individual treatment centers. The SEER (Surveillance, Epidemiology and End Results) database is a validated national epidemiological surveillance system and cancer registry that has been used extensively to evaluate treatment outcomes in cases of malignancy. The purpose of the present study was to use this database to identify demographic and prognostic characteristics of chondrosarcoma and to describe the natural history following the treatment of this rare disease in the United States over the last thirty years. Two thousand eight hundred and ninety patients with chondrosarcoma were identified in the SEER database, and information regarding the demographic and clinical characteristics of the patients, the histological features and grade of the tumors, the location and size of the tumors, the surgical stage at the time of diagnosis, the use of surgery and radiation treatment, and survival were extracted. Comparison of the overall and disease-specific survival rates revealed that patients who survived for ten years were more likely to die of events that were unrelated to chondrosarcoma. The disease-specific survival rate leveled off at ten years of follow-up. Univariate analysis revealed that female sex, a low histological grade, and local surgical stage were associated with a significant disease-specific survival benefit. An age of fifty years or less and an appendicular location of the tumor were associated with a significant overall survival benefit. On multivariate analysis, only grade and stage had significant association with disease-specific survival. On the basis of a comparison of survival rates according to the decade of diagnosis, it appears that there has been no significant improvement in survival over the last thirty years. Only grade and stage are independent prognostic factors for survival in cases of chondrosarcoma. Current treatment algorithms have not improved the survival rates of patients with chondrosarcoma over the past thirty years. Routine patient surveillance following treatment should be extended to ten years of follow-up.

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Author and article information

Contributors

Takashi Fukushima: d1617@jichi.ac.jp

Koichi Ogura: ogura-tky@umin.ac.jp

Toru Akiyama:

ORCID: http://orcid.org/0000-0001-9493-4768

toruakiyama827@jichi.ac.jp

Katsushi Takeshita: dtstake@jichi.ac.jp

Akira Kawai: akawai@ncc.go.jp

Journal

Journal ID (nlm-ta): BMC Musculoskelet Disord

Journal ID (iso-abbrev): BMC Musculoskelet Disord

Title: BMC Musculoskeletal Disorders

Publisher: BioMed Central (London )

ISSN (Electronic): 1471-2474

Publication date (Electronic): 18 August 2018

Publication date PMC-release: 18 August 2018

Publication date Collection: 2018

Volume: 19

Electronic Location Identifier: 297

Affiliations

[1 ]ISNI 0000000123090000, GRID grid.410804.9, Department of Orthopaedic Surgery, Saitama Medical Center, , Jichi Medical University, ; Saitama, Japan

[2 ]ISNI 0000000123090000, GRID grid.410804.9, Department of Orthopaedic Surgery, , Jichi Medical University, ; Tochigi, Japan

[3 ]ISNI 0000 0001 2168 5385, GRID grid.272242.3, Department of Musculoskeletal Oncology, , National Cancer Center Hospital, ; Tokyo, Japan

[4 ]ISNI 0000 0001 2151 536X, GRID grid.26999.3d, Department of Orthopaedic Surgery, Faculty of Medicine, , The University of Tokyo, ; Tokyo, Japan

Author information

Toru Akiyama http://orcid.org/0000-0001-9493-4768

Article

Publisher ID: 2217

DOI: 10.1186/s12891-018-2217-1

PMC ID: 6098838

PubMed ID: 30121085

SO-VID: daba62de-d7bb-4abf-8bd4-155ecf0833cd

License:

Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

History

Date received : 9 January 2018

Date accepted : 1 August 2018

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ScienceOpen disciplines: Orthopedics

Keywords: bone sarcoma,adolescent and young adult,cancer survival,japan,database

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ScienceOpen disciplines: Orthopedics

Keywords: bone sarcoma, adolescent and young adult, cancer survival, japan, database

Descriptive epidemiology and outcomes of bone sarcomas in adolescent and young adult patients in Japan

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Most cited references 21

Descriptive epidemiology of sarcomas in Europe: Report from the RARECARE project

The distinctive biology of cancer in adolescents and young adults.

Chondrosarcoma in the United States (1973 to 2003): an analysis of 2890 cases from the SEER database.

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