Pain, negative health perceptions, and certain coping styles for dealing with daily stressors are all linked to a greater incidence of depression among people with Fabry disease, according to a recent study.
The study, “Depressive symptoms in Fabry disease: the importance of coping, subjective health perception and pain,” was published in the Orphanet Journal of Rare Disease.
Compared with the general population, a high prevalence of depressive symptoms has been reported in individuals with Fabry.
Although it is not clear which specific characteristics may directly relate to the development of such symptoms, earlier studies have identified certain risk factors for depression in this patient population. Specifically, these include pain, patients’ perceptions of their health, being single, and a lack of social support.
An underlying biological cause relating to the cerebral pathology of Fabry disease also has been associated with the development of depression, although most studies have yet to validate a relation between organ involvement and depressive symptoms.
Determining how different factors relate to the depressive symptoms experienced by people with Fabry is important, as it can help identify those at risk while guiding both prevention and treatment of these symptoms.
Now, researchers from the Amsterdam University Medical Center, in the Netherlands, sought to identify which variables related to depressive symptoms in Fabry. The team also evaluated which coping styles — cognitive and behavioral efforts to control stresses and daily hassles — are most used by this patient population in relation to these symptoms.
A total of 81 individuals from the Dutch Fabry cohort at the Academic Medical Center were screened by the scientists. All of the patients had filled out questionnaires and completed a comprehensive neuropsychological assessment.
The participants’ mean age was 44.5 years and 28 (34.6%) were men. The majority (60 patients, 74.1%) had classical Fabry disease, and 43 (53.1%) were being treated with enzyme replacement therapy. A history of, or current depression was reported by 22 patients (27.2%).
Depressive symptoms were assessed using the Center for Epidemiologic Studies Depression (CESD) scale, which grades 20 items on a 0-to-3 scale, for a total score between 0 and 60 points. Scores equal to or greater than 16 indicate the presence of depressive symptoms, with higher scores indicating worse depression.
The Utrecht Coping List, a 47-item questionnaire that measures the use of seven different coping styles, was employed to assess each participant. The coping styles measured in this test are palliative, passive, active, avoiding, social support seeking, reassuring thoughts, and expressing emotions. Each response is rated from 1, indicating never, to 4, for often. The scores are compiled for each coping style, with higher scores indicating which is used most often.
Pain was measured using the Brief Pain Inventory (BPI), with scores ranging from 0 — no pain — to 10 for the worst possible pain. Quality of life was assessed using the 36-item Short Form Health Survey (SF-36), with higher scores indicating better functioning.
The Pittsburgh Sleep Quality Index (PSQI) was used to assess the quality of the participants’ sleep. The PSQI uses a 0-to-21 scale, with scores greater than five indicating poor sleep quality.
The researchers also performed a search for published studies to identify variables related to depressive symptoms in Fabry. A total of 16 studies were selected that identified six potential factors significantly linked to depression in early Fabry disease: pain, unemployment, health perception, being single, comorbidities or other concurrent medical conditions, and a history of stroke.
The results showed that 31 of the 81 patients (38%) had depressive symptoms and 13 (16%) had objective cognitive impairment.
The analysis identified three coping styles: “avoidance and brooding,” “positivity and problem solving,” and “seeking social support.”
Using three different statistical models, the researchers evaluated the link between the different factors and depression.
In one model, the team assessed the impact of the six potential factors identified in the literature in relation to depression. In the second model, they added the influence of the coping styles. The impact of additional factors was evaluated in the third model.
Higher depressive symptom scores were found for those patients with more pain and an “avoidance and brooding” coping style. Meanwhile, individuals with a better health perception and a more “positivity and problem solving” coping style had lower depressive symptom scores.
No relationship between unemployment and depressive symptoms was identified in these models. There also was no link between objective cognitive impairment and depressive symptoms. Further, the duration of treatment with enzyme replacement therapy was not found to be related to depressive symptoms.
Quality of life, loneliness, and cardiac or renal involvement explained most of the variability in depressive symptom scores.
“Depressive symptoms are frequent in patients with FD [Fabry disease] and are related to pain, negative health perception and use of specific coping styles,” the researchers wrote.
“Future psychological treatment can be tailored to coping styles, for example by focusing on improvement of problem solving or decreasing avoidant behavior, ideally in a research setting,” they added.
The researchers also recommended that pain should be routinely assessed, and that a screening questionnaire to detect depression should be added to routine clinical care.
“Patients with depressive symptoms should be referred, preferentially to psychologists with knowledge of chronic diseases,” they concluded.
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