Psychological resilience in patients with Friedreich ataxia: a 6-year longitudinal analysis.

BACKGROUND

Friedreich ataxia is a debilitating multisystem neurodegenerative disorder. Depression is more prevalent in individuals with Friedreich ataxia (14%-36%) than in the general European population (8.5%). While psychological resilience is known to influence mental health in other chronic conditions, its role in Friedreich ataxia remains underexplored.

OBJECTIVES

We aimed to compare psychological resilience between individuals with Friedreich ataxia and healthy controls, assess associations with clinical or demographic characteristics, depression, and anxiety, and explore changes in resilience longitudinally.

METHODS

Participants were drawn from the Aachen site of the European Friedreich Ataxia Consortium for Translational Studies (EFACTS) registry. Resilience was measured using the Wagnild and Young RS-25 questionnaire. Depression and anxiety were assessed using the Hospital Anxiety and Depression Scale and Beck's Depression Inventory II. Student's t-tests were used to compare groups. Pearson correlations assessed associations between resilience, depression, and anxiety scores. Univariate and multiple linear regressions were conducted to identify factors associated with resilience. Longitudinal resilience trends were evaluated graphically, and a within-person analysis was conducted using paired-T-tests in participants with available data. Missing data were imputed in 10% of cases.

RESULTS

Overall, 61 participants (43 with Friedreich ataxia, 18 controls) were followed for up to six years. Median age was 30 years for individuals with Friedreich ataxia and 28 years for controls; 58.1% and 61.1% were female, respectively. At baseline, resilience scores were significantly lower in individuals with Friedreich ataxia (135.1 ± 25.3) than in controls (151.5 ± 12.2; p < 0.05). Resilience was strongly negatively correlated with depression (r = -0.85, p < 0.001) and moderately with anxiety (r = -0.65, p < 0.001). We observed an apparent decline in resilience in patients during the COVID-19 pandemic albeit not statistically significant in the within-person subgroup analysis (pre-COVID-19 143.1 vs 131.2 post COVID-19; p = 0.07, n = 18), possibly due to low sample size.

CONCLUSIONS

Individuals with Friedreich ataxia display reduced resilience compared to healthy controls, which is associated with greater levels of depression and anxiety. Resilience remained stable over the disease course but appeared to decline during the COVID-19 pandemic, underlining the importance of strengthening resilience and mental well-being in this population.