Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
. 2025 Apr 15:13:1534153.
doi: 10.3389/fped.2025.1534153. eCollection 2025.

A multidisciplinary and structured approach for comprehensive evaluation of functional outcomes, adverse events, psychosocial outcomes and health-related quality of life after local therapy for bone sarcoma in children: protocol for a cross-sectional study

Leonie G Tigelaar  1 , Lianne M Haveman  1 , Willem P Bekkering  1 , Irene L B Oude Lansink  2 , Christel D Rohrich  2 , Hinke Van der Hoek  1 , Laura R Beek  1 , Jennifer Van Dijk  1 , Marjolein E M Langemeijer  1 , Relinde W Slooff-Lentink  1 , Alied M Van der Aa-Van Delden  1 , Heleen Maurice-Stam  1 , Annemarie M L Peek  1 , Helena J H Van der Pal  1 , Maria M W Koopman  1 , Leontien C M Kremer  1 , Stijn J Westerbos  1 , Harm Van Tinteren  1 , Jos A M Bramer  1   3 , Michiel A J Van de Sande  1   4 , Martha A Grootenhuis  1 , Hendrik W B Schreuder  1   5 , Johannes H M Merks  1   6
Affiliations

A multidisciplinary and structured approach for comprehensive evaluation of functional outcomes, adverse events, psychosocial outcomes and health-related quality of life after local therapy for bone sarcoma in children: protocol for a cross-sectional study

Leonie G Tigelaar et al. Front Pediatr. .

Abstract

Introduction: Bone sarcoma patients face intensive treatment, including life-changing local therapy, which impacts both short- and long-term functioning. Moreover, bone sarcoma survivors experience the highest burden of adverse events of all childhood cancer survivors. To address these issues, we set up a structured multidisciplinary outpatient follow-up clinic for patients who completed treatment and integrated this clinic into the standard of care. This study protocol describes the methodology of a cross-sectional study that aims to systematically report the functional outcomes, adverse events, psychosocial outcomes and health-related quality of life of the cohort seen at this clinic.

Methods and analysis: Participants are recruited at the multidisciplinary follow-up clinic and their consent is obtained. Standard of care clinical assessments serve as the primary data source for this study. Furthermore, additional research assessments are performed to further expand our knowledge. Assessments are structured by standardized assessment sets that we developed based on literature review and joint national expertise in bone sarcoma care. The sets comply with international guidelines such as the World Health Organization's International Classification of Functioning, disability and health, and include a combination of patient-reported, clinician-reported and performance-based outcome measures for comprehensive representation of outcomes.

Discussion: This study will generate valuable knowledge on the functional outcomes, adverse events, psychosocial outcomes and quality of life of a national cohort of pediatric bone sarcoma patients in follow-up care. By aligning additional research assessments with standardized patient care, a comprehensive range of outcomes will be obtained while minimizing the patient's burden. Moreover, this protocol may serve as a template for clinics and research internationally, allowing for the merging of standardized outcome data in such rare disease. This will facilitate the optimization of current patient care and inform the important shared decision-making process for local treatment in future patients.

Keywords: adverse events; bone sarcoma; functional outcome; orthopedic surgery; pediatric oncology; quality of life; study protocol.

PubMed Disclaimer

Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest

Figures

Figure 1
Figure 1
Informed consent process.
Figure 2
Figure 2
Setup of multidisciplinary clinic including incorporation of research assessments in italics.
Figure 3
Figure 3
International classification of functioning, disability and health. Original work by “World Health Organization (7). License: CC BY-NC-SA 3.0 IGO”.
See this image and copyright information in PMC

References

    1. Dutch Childhood Oncology Group Registry. Available at: https://www.skion.nl/info/4208/kinderoncologie-in-cijfers/4209/skion-bas... (Accessed September 29, 2023).
    1. Geenen MM, Cardous-Ubbink MC, Kremer LCM, Van Den Bos C, Van Der Pal HJH, Heinen RC, et al. Medical assessment of adverse health outcomes in long-term survivors of childhood cancer. J Am Med Assoc. (2007) 297:2705–15. 10.1001/jama.297.24.2705 - DOI - PubMed
    1. Stokke J, Sung L, Gupta A, Lindberg A, Rosenberg AR. Systematic review and meta-analysis of objective and subjective quality of life among pediatric, adolescent, and young adult bone tumor survivors. Pediatr Blood Cancer. (2015) 62:1616–29. 10.1002/pbc.25514 - DOI - PMC - PubMed
    1. Whelan JS, Bielack SS, Marina N, Smeland S, Jovic G, Hook JM, et al. EURAMOS-1, an international randomised study for osteosarcoma: results from pre-randomisation treatment. Ann Oncol. (2015) 26:407–14. 10.1093/annonc/mdu526 - DOI - PMC - PubMed
    1. Anderton J, Moroz V, Marec-Bérard P, Gaspar N, Laurence V, Martín-Broto J, et al. International randomised controlled trial for the treatment of newly diagnosed EWING sarcoma family of tumours—EURO EWING 2012 protocol. Trials. (2020) 21:1–9. 10.1186/s13063-019-4026-8 - DOI - PMC - PubMed

LinkOut - more resources