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18-03-2025

Family functioning and health-related quality of life in children and young adults with Marfan syndrome

Auteurs: Lauren S. Crafts, Lynn A. Sleeper, Karen Uzark, Andrew M. Atz, Timothy J. Bradley, Elizabeth Cappella, Veronica Fettig, Georgeann Keh Teng Groh, Michelle S. Hamstra, Christina Hartje-Dunn, Jami C. Levine, Ian Lindsay, Aaron K. Olson, Gretchen MacCarrick, Larry W. Markham, Tonia Morrison, Reed E. Pyeritz, Seda Tierney, Ronald V. Lacro

Gepubliceerd in: Quality of Life Research

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Abstract

Purpose

Evaluate family functioning (FF) and associations with quality of life (QOL) in a large, multicenter cohort of children and young adults with Marfan syndrome (MFS) who participated in the Pediatric Heart Network (PHN) Marfan Trial.

Methods

Of the 608 patients enrolled in the PHN Marfan Trial, 359 families completed one or more of the following: the General Functioning subscale of the Family Assessment Device (FAD), the Condition Management Ability module of the Family Management Measure (FaMM), and the Pediatric Quality of Life Inventory (PedsQL) at the final trial visit at three years. The correlations between FAD and FaMM scores and patient-related factors were examined. Linear regression was used to determine the relationship between FAD, FaMM, patient-related factors, and PedsQL.

Results

25% of families by self-report and 9% of families by parent-report had impaired functioning (FAD score > 2). FAD and FaMM were inversely correlated (R=-0.41, p < 0.001). Higher (worse) FAD scores were correlated with more patient-reported symptoms (R = 0.17, p = 0.003) and with lower QOL (R=-0.28, p < 0.001), independent of other factors typically related to QOL.

Conclusions

In patients with MFS, family dysfunction and more symptoms were independently associated with worse QOL. This suggests that interventions addressing family dysfunction and symptoms may positively impact QOL for patients and families living with MFS.
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Metagegevens
Titel
Family functioning and health-related quality of life in children and young adults with Marfan syndrome
Auteurs
Lauren S. Crafts
Lynn A. Sleeper
Karen Uzark
Andrew M. Atz
Timothy J. Bradley
Elizabeth Cappella
Veronica Fettig
Georgeann Keh Teng Groh
Michelle S. Hamstra
Christina Hartje-Dunn
Jami C. Levine
Ian Lindsay
Aaron K. Olson
Gretchen MacCarrick
Larry W. Markham
Tonia Morrison
Reed E. Pyeritz
Seda Tierney
Ronald V. Lacro
Publicatiedatum
18-03-2025
Uitgeverij
Springer New York
Gepubliceerd in
Quality of Life Research
Print ISSN: 0962-9343
Elektronisch ISSN: 1573-2649
DOI
https://doi.org/10.1007/s11136-025-03944-9