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Impact of childhood leukemia on siblings: their long-term perception of family functioning and its relationship with their psychosocial characteristics using structural equation modeling

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Abstract

Purpose

In the context of pediatric cancer, siblings’ adaptation and needs have been previously investigated; however, research on the long-term consequences on siblings, especially on their family environment, is scarce.

We aimed to (1) assess the family functioning (FF) perceived by siblings of childhood leukemia survivors long after diagnosis and (2) explore characteristics likely associated and investigate associations with psycho-behavioral and social factors.

Methods

Childhood leukemia survivors’ siblings older than 11 years were recruited through the LEA cohort, a French long-term follow-up program, and completed the family assessment device (FAD). Logistic regression analysis was used to determine factors likely associated with unhealthy functioning in families as perceived by siblings. Structural equation modeling (SEM) was used to examine relationships that predict siblings’ perception of FF.

Results

We included 605 siblings (mean follow-up time from diagnosis 14.1 ± 6.8 years), of whom 175 (28.9%) perceived unhealthy functioning. SEM showed that older siblings were more likely to perceive problematic functioning (β = 0.095, p = 0.014). Sex and leukemia burden had indirect effects on FF through mediators. Family financial situation at diagnosis was not associated with the risk of reporting unhealthy functioning.

Conclusions

Our study contributed to identifying siblings at risk of facing family issues and reinforced the need to provide more consideration and suitable resources to avoid late consequences. Often considered as the “forgotten children”, future research should focus on developing targeted interventions to facilitate family communication and improve siblings’ social support.

Implications for cancer survivors

Overall, results regarding FF perceived by siblings are reassuring and provide new enlightening elements that allow for better support to all families.

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Data availability

The datasets analyzed during the current study are available from the corresponding author on reasonable request.

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Acknowledgements

The authors thank all of the survivors, their families, and their siblings who agreed to participate in this study. The authors gratefully acknowledge the entire LEA Study Group (supplementary file 2).

Funding

This work was supported by grants from the French National Clinical Research Program (PHRC-N), the French National Cancer Institute (INCa) and the Assistance Publique of Hôpitaux de Marseille.

Author information

Authors and Affiliations

  1. UR 3279, CERESS – Health Service Research and Quality of Life Center, Aix-Marseille University, Marseille, France

    Cindy Faust, Pascal Auquier, Gérard Michel & Julie Berbis

  2. Department of Pediatric Hematology-Oncology, University Hospital of Lyon, Lyon, France

    Yves Bertrand

  3. Department of Pediatric Hematology-Oncology, A. Trousseau Hospital, AP-HP, Paris, France

    Marie-Dominique Tabone

  4. Department of Pediatric Hematology-Oncology, University Hospital of Bordeaux, Bordeaux, France

    Sophie Ansoborlo

  5. Department of Pediatric Hematology-Oncology, Saint Louis Hospital, AP-HP, Paris, France

    André Baruchel

  6. Department of Pediatric Hematology-Oncology, University Hospital of Rennes, Rennes, France

    Virginie Gandemer

  7. Department of Pediatric Hematology, Robert Debré Hospital, AP-HP, Paris, France

    Jean-Hugues Dalle

  8. Department of Pediatric Hematology-Oncology, Children’s Hospital of Brabois, Vandoeuvre Les Nancy, France

    Pascal Chastagner

  9. Department of Pediatric Hematology-Oncology, University Hospital of Clermont-Ferrand, CIC Inserm 501, Clermont-Ferrand, France

    Justyna Kanold

  10. Department of Pediatric Hematology-Oncology, University Hospital of Nice, Nice, France

    Maryline Poirée

  11. Department of Pediatric Hematology-Oncology, University Hospital of Montpellier, Montpellier, France

    Alexandre Theron

  12. Department of Pediatric Hematology-Oncology, University Hospital of Toulouse, Toulouse, France

    Geneviève Plat

  13. Department of Pediatric Hematology-Oncology, University Hospital of Angers, Angers, France

    Isabelle Pellier

  14. Department of Pediatric Hematology-Oncology, Timone Children’s Hospital and Aix-Marseille University, Marseille, France

    Gérard Michel

Authors
  1. Cindy Faust
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  2. Pascal Auquier
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  3. Yves Bertrand
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  4. Marie-Dominique Tabone
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  5. Sophie Ansoborlo
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  15. Gérard Michel
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  16. Julie Berbis
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Contributions

Cindy Faust: formal analysis, funding acquisition, methodology, visualization, writing—original draft, and writing—review and editing. Pascal Auquier: conceptualization, funding acquisition, supervision, validation, and writing—review and editing. Yves Bertrand, Marie-Dominique Tabone, Sophie Ansoborlo, André Baruchel, Virginie Gandemer, Jean-Hugues Dalle, Aurélie Phulpin, Justyna Kanold, Maryline Poirée, Alexandre Theron, Geneviève Plat, Isabelle Pellier: funding acquisition, investigation, resources, and writing—review and editing. Gérard Michel: conceptualization, funding acquisition, investigation, resources, and writing—review and editing. Julie Berbis: funding acquisition, methodology, supervision, validation, and writing—review and editing.

Corresponding author

Correspondence to Cindy Faust.

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Ethics approval

This study was performed in line with the principles of the Declaration of Helsinki. Approval was granted by the Ethics Committee SUD MÉDITERRANÉE (reference RO–2016/20).

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This is an observational study. All participants received an information note and had the right to withdraw consent at any time.

Competing interests

The authors declare no competing interests.

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Faust, C., Auquier, P., Bertrand, Y. et al. Impact of childhood leukemia on siblings: their long-term perception of family functioning and its relationship with their psychosocial characteristics using structural equation modeling. J Cancer Surviv (2023). https://doi.org/10.1007/s11764-023-01385-2

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