Marielle Bassel

Psychological health and well-being in systemic sclerosis: State of the science and consensus research agenda.

Introduction Systemic sclerosis (SSc; scleroderma) is a multisystem disorder characterized by disturbance in fibroblast function, microvascular disease, and immune system activation, culminating in fibrosis of the skin and internal organs (1,2). SSc is associated with extensive morbidity, including disfiguring skin thickening, finger ulcers, joint contractures, pulmonary hypertension, interstitial lung disease, chronic diarrhea, and renal failure (1,2). The rate of disease onset is highest between 30 and 50 years of age, with the risk for women being 4 to 5 times higher than for men (3,4). Median survival time from diagnosis is 11 years, and patients are 3.7 times more likely to die within 10 years of diagnosis (44.9% mortality) than age-, sex-, and race-matched individuals without SSc (12.0% mortality) (3).

Sociodemographic, disease, and symptom correlates of fatigue in systemic sclerosis: Evidence from a sample of 659 Canadian Scleroderma Research Group Registry patients

OBJECTIVE To assess fatigue levels and demographic, socioeconomic, disease, and psychosocial correlates of fatigue in patients with systemic sclerosis (SSc). METHODS We conducted a cross-sectional, multicenter study of 659 patients with SSc from the Canadian Scleroderma Research Group Registry. Fatigue was assessed during annual Registry visits with the Short Form 36 (SF-36) health survey vitality subscale. Patients completed measures of depressive symptoms and pain and underwent clinical histories and medical examinations. Kendalls tau was used to assess bivariate association of sociodemographic, medical, and psychosocial variables with fatigue. Multivariable associations of demographic (step 1), socioeconomic (step 2), global disease (step 3), specific disease and lifestyle (step 4), and psychosocial (step 5) factors with fatigue were assessed using hierarchical multiple linear regression. RESULTS The mean +/- SD score of the patients on the SF-36 vitality subscale was 45.6 +/- 10.8, substantially lower (indicating more fatigue) than the mean +/- SD score for the Canadian general population (65.8 +/- 18.0). In multivariate analysis, higher fatigue was significantly associated with the number of medical comorbidities (standardized beta = -0.11, P = 0.004), breathing problems (standardized beta = -0.23, P < 0.001), the number of gastrointestinal (GI) symptoms (standardized beta = -0.27, P < 0.001), and current smoking (standardized beta = -0.08, P = 0.018). As a group, specific symptom and lifestyle variables predicted the most incremental variance in fatigue (R(2) = 21.6%, P < 0.001), despite being added to the model after demographic, socioeconomic, and global disease duration/severity indicators. Symptoms of depression (beta = -0.42) and pain (beta = -0.21) were also independently associated with fatigue (P < 0.001). CONCLUSION High levels of fatigue are common in patients with SSc and are independently associated with clinical variables, including number of comorbidities, breathing problems, GI symptoms, and smoking.

Is longer-term psychodynamic psychotherapy more effective than shorter-term therapies? Review and critique of the evidence.

Background: In 2008, Leichsenring and Rabung performed a meta-analysis of 8 studies of longer-term psychodynamic psychotherapy (LTPP). The work was published in the Journal of the American Medical Association (vol. 300, pp 1551–1565), and they concluded that LTPP was more effective than shorter-term therapies. Method: Given that such claims have the potential to influence treatment decisions and policies, we re-examined the meta-analysis and the 8 studies. Results: We found a miscalculation of the effect sizes used to make key comparisons. Claims for the effectiveness of LTPP depended on a set of small, underpowered studies that were highly heterogeneous in terms of patients treated, interventions, comparison-control groups, and outcomes. LTPP was compared to 12 types of comparison-controls, including control groups that did not involve any psychotherapy, short-term psychodynamic psychotherapy, and unvalidated treatments. Additionally, the studies failed to protect against threats to bias, and had poor internal validity. Conclusion: Overall, we found no evidence to support claims of superiority of LTPP over shorter-term methods of psychotherapy.

Association of pruritus with quality of life and disability in systemic sclerosis

To our knowledge, no studies have investigated the association of pruritus, which is present in almost half of patients with systemic sclerosis (SSc; scleroderma), with quality of life (QOL) and disability. The objective of this study was to investigate the association of pruritus with QOL and disability in SSc.

Prevalence and clinical correlates of pruritus in patients with systemic sclerosis.

OBJECTIVE There are no studies of pruritus prevalence or clinical correlates in systemic sclerosis (SSc). The objectives of this study were to document the proportion of SSc patients with pruritus on most days, to determine when in the course of the disease pruritus is most prevalent, and to identify clinical correlates. METHODS We performed a cross-sectional, multicenter study of 400 SSc patients from the Canadian Scleroderma Research Group Registry > or =1 year after Registry enrollment. Patients indicated whether they experienced pruritus during the past month on most days and underwent clinical histories and medical examinations. Multiple logistic regression was used to assess the association between sociodemographic and clinical variables and pruritus. RESULTS A total of 179 patients (45%) reported pruritus on most days, including 69% (11 of 16) among patients 1.0-1.9 years from onset of non-Raynauds symptoms, 41% (38 of 93) for 2.0-4.9 years, 47% (44 of 94) for 5.0-9.9 years, 43% (60 of 140) for 10.0-19.9 years, and 46% (26 of 57) for > or =20 years. In post hoc analysis, patients 1.0-1.9 years from disease onset were significantly more likely to report pruritus (P = 0.049). Patients with pruritus had significantly more skin involvement (P = 0.029), more gastrointestinal (GI) symptoms (P < 0.001), worse breathing problems (P = 0.001), worse Raynauds symptoms (P = 0.002), and more severe finger ulcers (P = 0.009). Only the number of GI symptoms predicted pruritus in multiple logistic regression analysis (odds ratio 1.25, 95% confidence interval 1.13-1.37; P < 0.001). CONCLUSION Pruritus is common in SSc and is independently associated with GI symptoms. Focused research on sources of pruritus and its management in SSc is needed.

Analyzing Effectiveness of Long-term Psychodynamic Psychotherapy

To the Editor: Drs Leichsenring and Rabung reported that long-term psychodynamic psychotherapy (LTPP) is more effective than shorter forms of psychotherapy for complex mental disorders based on a between-group effect size of 1.8 from 7 comparative trials that they meta-analyzed. The authors did not indicate that they were concerned about this and other surprisingly large effect sizes they reported. Between-group effect sizes can be presented as group differences in terms of standard deviations or as point biserial correlations between group (eg, LTPP vs shorter-term therapies) and treatment effect. They are equivalent and convertible using a formula or tables. The authors, however, apparently erroneously calculated within-group pre-post effect sizes and point biserial correlations between group and within-group effect sizes, which is altogether different. It seems that they converted these correlations between group and within-group pre-post effect sizes to produce deviation-based effect sizes that do not appear reasonable. As a result, although none of the 7 studies had an overall effect size greater than 1.45, the authors reported a combined effect size of 1.8, which is statistically impossible. In a slightly larger set of 8 trials, the authors reported that LTPP had a larger overall effect than shorter-term therapies (0.96 vs 0.47) but a point biserial correlation (0.60) equivalent to a between-group effect size of 1.5. However, betweengroup effect sizes must be smaller than within-group effect sizes when both groups have positive effects. Similarly, these methods generated an implausible between-group effect size of 6.9 for personality functioning based on 3 trials, none of which reported an effect size for personality functioning larger than approximately 2. In addition, we believe that this collection of studies was not suitable for meta-analysis. Each reviewed study had 15 to 30 patients in the LTPP treatment group. It seems unlikely that investigators would attempt to publish a negative study with so few patients (or that such a study would be accepted for publication), which means that all published studies would have an effect size of at least 0.50 to 0.75, the minimum for statistical significance. This is an artificial floor that guarantees a large effect when these studies are combined.

Sexual function in women with systemic sclerosis: Comment on the article by Schouffoer et al.

In an article published recently in Arthritis Care & Research, Schouffoer et al reported that women with systemic sclerosis (SSc; scleroderma) have greater sexual impairment and distress than healthy controls, and the authors recommended that health care professionals inquire about sexuality with all SSc patients (1). Two previous studies reported similarly that women with SSc have greater sexual impairment than women in the general population (2) or compared with women with other medical diseases where sexual problems are more routinely addressed (3). The broad recommendation of Schouffoer et al for routine inquiry about sexual problems, however, appears to be premature. Only 16% of patients in the study by Schouffoer et al expressed a desire to discuss sexual problems, and none wished to do this with their rheumatologist. While there is a great need to better address sexuality in SSc, it is important to demonstrate the patient benefits of routine inquiry before a specific recommendation is made. A call for routine inquiry about sexuality should only be made once we have provided a way of doing this that is agreeable to patients, feasibly implemented by physicians, and linked to an effective intervention and patient benefit (4). In order to develop and test an intervention, it is also important that we understand which disease and psychosocial characteristics may lead to sexual dysfunction. Schouffoer et al reported that “none of the specific disease characteristics of SSc were found to be associated with sexual problems” (1). Previous studies, however, have found that women with diffuse SSc have significantly greater impairment than those with limited SSc (3) and that women with SSc are more likely to experience dyspareunia, vaginal dryness, and vaginal ulcerations/fissures than women with other rheumatic diseases (5). The study by Schouffoer et al did not include an assessment of key SSc variables that may be related to impaired sexual function. Furthermore, based on the study’s small sample size, there was only 32% power to detect a moderate difference in sexual impairment between limited and diffuse patients (e.g., 0.50). Studies are needed that investigate multiple predictors of sexual impairment, including both physical symptoms of SSc and psychological factors, with sufficient power to detect potential upstream factors. The study by Schouffoer and colleagues is a step in the right direction and contributes to the limited body of research on sexual function in SSc. However, more research is needed before broad recommendations to routinely inquire about sexuality can be made. Before we ask health providers to reach beyond their level of comfort and training to address sexual issues, we need to provide them with a way to do it effectively. In the interim, patients may benefit from a referral to a specialist, as Schouffoer et al suggest, or from the provision of information (e.g., in the form of a pamphlet) about common SSc sexual issues, which may normalize the subject and facilitate discussion with a health professional (3). Ms Knafo and Ms Jewett are supported by Frederick Banting and Charles Best Canadian Graduate Scholarship/Master’s Awards from the Canadian Institutes of Health Research (CIHR) and by Canadian Scleroderma Research Group Studentships (CIHR Strategic Training Initiative in Health Research Grant). Dr. Thombs is supported by a New Investigator Award from the CIHR and an Établissement de Jeunes Chercheurs award from the Fonds de la Recherche en Santé Québec.