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. 2024 Feb 28:2:100053.
doi: 10.1016/j.hctj.2024.100053. eCollection 2024.

Wellbeing measurement among adults with Charcot-Marie-Tooth disease

Payton D Rule  1 , Megan W Wolk  1 , Patrick L Hill  1
Affiliations

Wellbeing measurement among adults with Charcot-Marie-Tooth disease

Payton D Rule et al. Health Care Transit. .

Abstract

Background and aims: Wellbeing research among individuals with Charcot-Marie-Tooth disease (CMT) is limited. The goal of the current study is to characterize the challenges experienced by adults with CMT that researchers may miss by utilizing typical strategies to capture mental health and wellbeing.

Methods: In 2021, we recruited 288 US adults with CMT ( M Age = 60 years, 59% Female, 93% White) to take part in an online survey. Participants were presented with validated wellbeing and health measures, in addition to newly created measures specific to CMT, and then asked to provide free-response feedback about aspects of their lived experiences missing from these measures. Thematic analysis were applied to the free-response data to identify CMT-related difficulties that may impact wellbeing among adults with CMT.

Results: Results identified nine CMT-related challenges that are important to individuals with CMT and are often left out of wellbeing assessments in this population. The three most common themes identified were anxiety or worry, coping with loss of function or progression, and difficulty finding knowledgeable medical providers. Further, some aspects of the CMT experience may be comorbid.

Interpretation: These results can be used to inform future research and clinical practice in the CMT population.

Keywords: Disabilities; Inherited peripheral neuropathy; Lived experience; Wellbeing.

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Conflict of interest statement

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

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References

    1. Hays R.D., Hahn H., Marshall G. Use of the SF-36 and other health-related quality of life measures to assess persons with disabilities. Arch Phys Med Rehabil. 2002;83:S4–S9. doi: 10.1053/apmr.2002.36837. - DOI - PubMed
    1. Carr A.J., Higginson I.J. Measuring quality of life: are quality of life measures patient centred? BMJ. 2001;322(7298):1357–1360. doi: 10.1136/bmj.322.7298.1357. - DOI - PMC - PubMed
    1. Albrecht G.L., Devlieger P.J. The disability paradox: high quality of life against all odds. Soc Sci Med. 1999;48(8):977–988. doi: 10.1016/S0277-9536(98)00411-0. - DOI - PubMed
    1. Smith M., Calder-Dawe O., Carroll P., et al. Mobility barriers and enablers and their implications for the wellbeing of disabled children and young people in Aotearoa New Zealand: a cross-sectional qualitative study. Wellbeing Space Soc. 2021;2 doi: 10.1016/j.wss.2021.100028. - DOI
    1. Ware J.E., Sherbourne C.D. The MOS 36-Ltem Short-form Health Survey (SF-36): I. Conceptual framework and item selection. Med Care. 1992;30(6):473–483. doi: 10.1097/00005650-199206000-00002. - DOI - PubMed

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