Secondary malignant neoplasms after bone and soft tissue sarcomas in children, adolescents, and young adults.


Journal

Cancer
ISSN: 1097-0142
Titre abrégé: Cancer
Pays: United States
ID NLM: 0374236

Informations de publication

Date de publication:
01 05 2022
Historique:
revised: 05 12 2021
received: 29 07 2021
accepted: 15 12 2021
pubmed: 24 2 2022
medline: 13 4 2022
entrez: 23 2 2022
Statut: ppublish

Résumé

Increased survival in young sarcoma patients comes along with a higher incidence of second malignant neoplasms (SMNs). The incidence, latency, histiotype, and outcome of these patients were analyzed because this information is essential to design evidence-based long-term follow-up care programs for young sarcoma survivors. Patients entered on clinical trials or registered in registries with a primary sarcoma in 1 of the cooperative sarcoma study groups in the framework of the Society for Pediatric Oncology and Hematology (GPOH) were screened for SMNs. Descriptive analysis, the Kaplan-Meier method, the Gray model, the Fine-Gray model, and the Cox regression model were used for the statistical analyses. A total of 159 out of 7079 (2.2%) patients were registered with a SMN. Among them, 104 solid SMNs (65%) and 56 hematologic SMNs (35%) occurred. Median latency from first diagnosis of sarcoma to the diagnosis of SMN was 6.8 years (range, 0-26.7 years). Cumulative incidence of SMN was 8.8% after 30 years. Five-year-survival was 67.1% (95% confidence interval [CI], 66.0-68.2) for the 7079 patients and it was 45.1% (95% CI, 36.2-53.6) after the diagnosis of a SMN (subcohort of n = 159 patients). There is a remarkable high cumulative incidence of SMNs after bone and soft tissue sarcomas in children, adolescents, and young adults. Therefore, effective transition as well as risk adapted long-term follow-up care programs should be developed and offered to young sarcoma survivors. Bone sarcomas and soft tissue tumors are rare tumors in children, adolescents, and young adults. The treatment varies, but may comprise chemotherapy, surgery, and/or radiotherapy. Developing a subsequent malignant tumor is a long-term risk for the patients. To better characterize this risk, we analyzed the data of 7079 patients (up to 21 years old) with bone sarcomas or soft tissue tumors. Our findings provide a basis to counsel young sarcoma survivors on their individual risk of subsequent malignant tumors. Moreover, these data can help to establish recommendations for aftercare in young sarcoma survivors.

Sections du résumé

BACKGROUND
Increased survival in young sarcoma patients comes along with a higher incidence of second malignant neoplasms (SMNs). The incidence, latency, histiotype, and outcome of these patients were analyzed because this information is essential to design evidence-based long-term follow-up care programs for young sarcoma survivors.
METHODS
Patients entered on clinical trials or registered in registries with a primary sarcoma in 1 of the cooperative sarcoma study groups in the framework of the Society for Pediatric Oncology and Hematology (GPOH) were screened for SMNs. Descriptive analysis, the Kaplan-Meier method, the Gray model, the Fine-Gray model, and the Cox regression model were used for the statistical analyses.
RESULTS
A total of 159 out of 7079 (2.2%) patients were registered with a SMN. Among them, 104 solid SMNs (65%) and 56 hematologic SMNs (35%) occurred. Median latency from first diagnosis of sarcoma to the diagnosis of SMN was 6.8 years (range, 0-26.7 years). Cumulative incidence of SMN was 8.8% after 30 years. Five-year-survival was 67.1% (95% confidence interval [CI], 66.0-68.2) for the 7079 patients and it was 45.1% (95% CI, 36.2-53.6) after the diagnosis of a SMN (subcohort of n = 159 patients).
CONCLUSIONS
There is a remarkable high cumulative incidence of SMNs after bone and soft tissue sarcomas in children, adolescents, and young adults. Therefore, effective transition as well as risk adapted long-term follow-up care programs should be developed and offered to young sarcoma survivors.
LAY SUMMARY
Bone sarcomas and soft tissue tumors are rare tumors in children, adolescents, and young adults. The treatment varies, but may comprise chemotherapy, surgery, and/or radiotherapy. Developing a subsequent malignant tumor is a long-term risk for the patients. To better characterize this risk, we analyzed the data of 7079 patients (up to 21 years old) with bone sarcomas or soft tissue tumors. Our findings provide a basis to counsel young sarcoma survivors on their individual risk of subsequent malignant tumors. Moreover, these data can help to establish recommendations for aftercare in young sarcoma survivors.

Identifiants

pubmed: 35195899
doi: 10.1002/cncr.34110
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

1787-1800

Informations de copyright

© 2022 American Cancer Society.

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Auteurs

Stefanie J Kube (SJ)

Pediatrics 1, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Claudia Blattmann (C)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Stefan S Bielack (SS)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Leo Kager (L)

Department of Pediatrics, St. Anna Children's Hospital, Medical University Vienna, Vienna, Austria.

Peter Kaatsch (P)

German Childhood Cancer Registry, Institute for Medical Biostatistics, Epidemiology and Informatics, University Medical Center of the Johannes Gutenberg-University Mainz, Mainz, Germany.

Thomas Kühne (T)

University Children's Hospital Basel, Basel, Switzerland.

Benjamin Sorg (B)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Matthias Kevric (M)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Susanne Jabar (S)

Cooperative Ewing Sarcoma Study Group, Essen University Hospital, Essen, Germany.

Erika Hallmen (E)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Monika Sparber-Sauer (M)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Thomas Klingebiel (T)

Division of Pediatric Hematology and Oncology, Hospital for Children and Adolescents, Johann Wolfgang Goethe University, Frankfurt, Germany.

Ewa Koscielniak (E)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.
University of Tuebingen, Tuebingen, Germany.

Uta Dirksen (U)

Cooperative Ewing Sarcoma Study Group, Essen University Hospital, Essen, Germany.

Stefanie Hecker-Nolting (S)

Pediatrics 5, Olgahospital, Klinikum Stuttgart, Stuttgart, Germany.

Joachim W O Gerß (JWO)

Institute of Biostatistics and Clinical Research, University of Münster, Münster, Germany.

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