Browsing by Author "Hudson, Marie"
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- ItemOpen AccessEffects of a support group leader education program jointly developed by health professionals and patients on peer leader self-efficacy among leaders of scleroderma support groups: a two-arm parallel partially nested randomised controlled trial(2022-10-28) Thombs, Brett D.; Levis, Brooke; Carrier, Marie-Eve; Dyas, Laura; Nordlund, Julia; Tao, Lydia; Aguila, Kylene; Bourgeault, Angelica; Konrad, Violet; Sauvé, Maureen; Connolly, Kerri; Henry, Richard S.; Østbø, Nora; Levis, Alexander W.; Kwakkenbos, Linda; Malcarne, Vanessa L.; El-Baalbaki, Ghassan; Hudson, Marie; Wurz, Amanda; Culos-Reed, S. N.; Platt, Robert W.; Benedetti, AndreaAbstract Background More people with rare diseases likely receive disease education and emotional and practical support from peer-led support groups than any other way. Most rare-disease support groups are delivered outside of the health care system by untrained leaders. Potential benefits may not be achieved and harms, such as dissemination of inaccurate information, may occur. Our primary objective was to evaluate the effects of a rare-disease support group leader education program, which was developed collaboratively by researchers, peer support group leaders, and patient organization leaders, compared to waitlist control, on peer leader self-efficacy among scleroderma support group leaders. Methods The trial was a pragmatic, two-arm partially nested randomised controlled trial with 1:1 allocation into intervention or waitlist control. Eligible participants were existing or candidate peer support group leaders affiliated with a scleroderma patient organization. Leader training was delivered in groups of 5–6 participants weekly for 13 weeks in 60–90 min sessions via the GoToMeeting® videoconferencing platform. The program included 12 general leader training modules and one module specific to scleroderma. Primary outcome was leader self-efficacy, measured by the Support Group Leader Self-efficacy Scale (SGLSS) immediately post-intervention. Secondary outcomes were leader self-efficacy 3 months post-intervention; emotional distress, leader burnout, and volunteer satisfaction post-intervention and 3 months post-intervention; and program satisfaction among intervention participants post-intervention. Results One hundred forty-eight participants were randomised to intervention (N = 74) or waitlist (N = 74). Primary outcome data were provided by 146 (99%) participants. Mean number of sessions attended was 11.4 (standard deviation = 2.6). Mean program satisfaction score (CSQ-8) was 30.3 (standard deviation = 3.0; possible range 8–32). Compared to waitlist control, leader self-efficacy was higher post-intervention [SGLSS; 16.7 points, 95% CI 11.0–22.3; standardized mean difference (SMD) 0.84] and 3 months later (15.6 points, 95% CI 10.2–21.0; SMD 0.73); leader volunteer satisfaction was significantly higher at both assessments, emotional distress was lower post-intervention but not 3 months later, and leader burnout was not significantly different at either assessment. Conclusions Peer support group leader education improved leader self-efficacy substantially. The program could be easily adapted for support group leaders in other rare diseases. Trial registration: NCT03965780 ; registered on May 29, 2019.
- ItemOpen AccessLong-term exposure to a mixture of industrial SO2, NO2, and PM2.5 and anti-citrullinated protein antibody positivity(2020-07-29) Zhao, Naizhuo; Smargiassi, Audrey; Hatzopoulou, Marianne; Colmegna, Ines; Hudson, Marie; Fritzler, Marvin J; Awadalla, Philip; Bernatsky, SashaAbstract Background Studies of associations between industrial air emissions and rheumatic diseases, or diseases-related serological biomarkers, are few. Moreover, previous evaluations typically studied individual (not mixed) emissions. We investigated associations between individual and combined exposures to industrial sulfur dioxide (SO2), nitrogen dioxide (NO2), and fine particles matter (PM2.5) on anti-citrullinated protein antibodies (ACPA), a characteristic biomarker for rheumatoid arthritis (RA). Methods Serum ACPA was determined for 7600 randomly selected CARTaGENE general population subjects in Quebec, Canada. Industrial SO2, NO2, and PM2.5 concentrations, estimated by the California Puff (CALPUFF) atmospheric dispersion model, were assigned based on residential postal codes at the time of sera collection. Single-exposure logistic regressions were performed for ACPA positivity defined by 20 U/ml, 40 U/ml, and 60 U/ml thresholds, adjusting for age, sex, French Canadian origin, smoking, and family income. Associations between regional overall PM2.5 exposure and ACPA positivity were also investigated. The associations between the combined three industrial exposures and the ACPA positivity were assessed by weighted quantile sum (WQS) regressions. Results Significant associations between individual industrial exposures and ACPA positivity defined by the 20 U/ml threshold were seen with single-exposure logistic regression models, for industrial emissions of PM2.5 (odds ratio, OR = 1.19, 95% confidence intervals, CI: 1.04–1.36) and SO2 (OR = 1.03, 95% CI: 1.00–1.06), without clear associations for NO2 (OR = 1.01, 95% CI: 0.86–1.17). Similar findings were seen for the 40 U/ml threshold, although at 60 U/ml, the results were very imprecise. The WQS model demonstrated a positive relationship between combined industrial exposures and ACPA positivity (OR = 1.36, 95% CI: 1.10–1.69 at 20 U/ml) and suggested that industrial PM2.5 may have a closer association with ACPA positivity than the other exposures. Again, similar findings were seen with the 40 U/ml threshold, though 60 U/ml results were imprecise. No clear association between ACPA and regional overall PM2.5 exposure was seen. Conclusions We noted positive associations between ACPA and industrial emissions of PM2.5 and SO2. Industrial PM2.5 exposure may play a particularly important role in this regard.
- ItemOpen AccessMyositis with prominent B-cell aggregates causing shrinking lung syndrome in systemic lupus erythematosus: a case report(2022-02-16) Roy, Flavie; Korathanakhun, Pat; Karamchandani, Jason; Dubé, Bruno-Pierre; Landon-Cardinal, Océane; Routhier, Nathalie; Peyronnard, Caroline; Massie, Rami; Leclair, Valérie; Meyer, Alain; Bourré-Tessier, Josiane; Satoh, Minoru; Fritzler, Marvin J.; Senécal, Jean-Luc; Hudson, Marie; O’Ferrall, Erin K.; Troyanov, Yves; Ellezam, Benjamin; Makhzoum, Jean-PaulAbstract Background Shrinking lung syndrome (SLS) is a rare manifestation of systemic lupus erythematosus (SLE) characterized by decreased lung volumes and diaphragmatic weakness in a dyspneic patient. Chest wall dysfunction secondary to pleuritis is the most commonly proposed cause. In this case report, we highlight a new potential mechanism of SLS in SLE, namely diaphragmatic weakness associated with myositis with CD20 positive B-cell aggregates. Case presentation A 51-year-old Caucasian woman was diagnosed with SLE and secondary Sjögren’s syndrome based on a history of pleuritis, constrictive pericarditis, polyarthritis, photosensitivity, alopecia, oral ulcers, xerophthalmia and xerostomia. Serologies were significant for positive antinuclear antibodies, anti-SSA, lupus anticoagulant and anti-cardiolopin. Blood work revealed a low C3 and C4, lymphopenia and thrombocytopenia. She was treated with with low-dose prednisone and remained in remission with oral hydroxychloroquine. Seven years later, she developed mild proximal muscle weakness and exertional dyspnea. Pulmonary function testing revealed a restrictive pattern with small lung volumes. Pulmonary imaging showed elevation of the right hemidiaphragm without evidence of interstitial lung disease. Diaphragmatic ultrasound was suggestive of profound diaphragmatic weakness and dysfunction. Based on these findings, a diagnosis of SLS was made. Her proximal muscle weakness was investigated, and creatine kinase (CK) levels were normal. Electromyography revealed fibrillation potentials in the biceps, iliopsoas, cervical and thoracic paraspinal muscles, and complex repetitive discharges in cervical paraspinal muscles. Biceps muscle biopsy revealed dense endomysial lymphocytic aggregates rich in CD20 positive B cells, perimysial fragmentation with plasma cell-rich perivascular infiltrates, diffuse sarcolemmal upregulation of class I MHC, perifascicular upregulation of class II MHC, and focal sarcolemmal deposition of C5b-9. Treatment with prednisone 15 mg/day and oral mycophenolate mofetil 2 g/day was initiated. Shortness of breath and proximal muscle weakness improved significantly. Conclusion Diaphragmatic weakness was the inaugural manifestation of myositis in this patient with SLE. The spectrum of myologic manifestations of myositis with prominent CD20 positive B-cell aggregates in SLE now includes normal CK levels and diaphragmatic involvement, in association with SLS.
- ItemOpen AccessRheumatoid arthritis-relevant DNA methylation changes identified in ACPA-positive asymptomatic individuals using methylome capture sequencing(2019-07-31) Shao, Xiaojian; Hudson, Marie; Colmegna, Ines; Greenwood, Celia M T; Fritzler, Marvin J; Awadalla, Philip; Pastinen, Tomi; Bernatsky, SashaAbstract Objective To compare DNA methylation in subjects positive vs negative for anti-citrullinated protein antibodies (ACPA), a key serological marker of rheumatoid arthritis (RA) risk. Methods With banked serum from a random subset (N = 3600) of a large general population cohort, we identified ACPA-positive samples and compared them to age- and sex-matched ACPA-negative controls. We used a custom-designed methylome panel to conduct targeted bisulfite sequencing of 5 million CpGs located in regulatory or hypomethylated regions of DNA from whole blood (red blood cell lysed). Using binomial regression models, we investigated the differentially methylated regions (DMRs) between ACPA-positive vs ACPA-negative subjects. An independent set of T cells from RA patients was used to “validate” the differentially methylated sites. Results We measured DNA methylation in 137 subjects, of whom 63 were ACPA-positive, 66 were ACPA-negative, and 8 had self-reported RA. We identified 1303 DMRs of relevance, of which one third (402) had underlying genetic effects. These DMRs were enriched in intergenic CpG islands (CGI) and CGI shore regions. Furthermore, the genes associated with these DMRs were enriched in pathways related to Epstein-Barr virus infection and immune response. In addition, 80 (38%) of 208 RA-specific DMRs were replicated in T cells from RA samples. Conclusions Sequencing-based high-resolution methylome mapping revealed biologically relevant DNA methylation changes in asymptomatic individuals positive for ACPA that overlap with those seen in RA. Pathway analyses suggested roles for viral infections, which may represent the effect of environmental triggers upstream of disease onset.
- ItemOpen AccessStatin-induced anti-HMGCR myopathy: successful therapeutic strategies for corticosteroid-free remission in 55 patients(2020-01-08) Meyer, Alain; Troyanov, Yves; Drouin, Julie; Oligny-Longpré, Geneviève; Landon-Cardinal, Océane; Hoa, Sabrina; Hervier, Baptiste; Bourré-Tessier, Josiane; Mansour, Anne-Marie; Hussein, Sara; Morin, Vincent; Rich, Eric; Goulet, Jean-Richard; Chartrand, Sandra; Hudson, Marie; Nehme, Jessica; Makhzoum, Jean-Paul; Zarka, Farah; Villeneuve, Edith; Raynauld, Jean-Pierre; Landry, Marianne; O’Ferrall, Erin K; Ferreira, Jose; Ellezam, Benjamin; Karamchandani, Jason; Larue, Sandrine; Massie, Rami; Isabelle, Catherine; Deschênes, Isabelle; Leclair, Valérie; Couture, Hélène; Targoff, Ira N; Fritzler, Marvin J; Senécal, Jean-LucAbstract Objective To describe successful therapeutic strategies in statin-induced anti-HMGCR myopathy. Methods Retrospective data from a cohort of 55 patients with statin-induced anti-HMGCR myopathy, sequentially stratified by the presence of proximal weakness, early remission, and corticosteroid and IVIG use at treatment induction, were analyzed for optimal successful induction and maintenance of remission strategies. Results A total of 14 patients achieved remission with a corticosteroid-free induction strategy (25%). In 41 patients treated with corticosteroids, only 4 patients (10%) failed an initial triple steroid/IVIG/steroid-sparing immunosuppressant (SSI) induction strategy. Delay in treatment initiation was independently associated with lower odds of successful maintenance with immunosuppressant monotherapy (OR 0.92, 95% CI 0.85 to 0.97, P = 0.015). While 22 patients (40%) presented with normal strength, only 9 had normal strength at initiation of treatment. Conclusion While corticosteroid-free treatment of anti-HMGCR myopathy is now a safe option in selected cases, initial triple steroid/IVIG/SSI was very efficacious in induction. Delays in treatment initiation and, as a corollary, delays in achieving remission decrease the odds of achieving successful maintenance with an SSI alone. Avoiding such delays, most notably in patients with normal strength, may reset the natural history of anti-HMGCR myopathy from a refractory entity to a treatable disease.
- ItemOpen AccessThe Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program: protocol for a two-arm parallel partially nested randomized controlled feasibility trial with progression to full-scale trial(2021-11-27) Nordlund, Julia; Henry, Richard S.; Kwakkenbos, Linda; Carrier, Marie-Eve; Levis, Brooke; Nielson, Warren R.; Bartlett, Susan J.; Dyas, Laura; Tao, Lydia; Fedoruk, Claire; Nielsen, Karen; Hudson, Marie; Pope, Janet; Frech, Tracy; Gholizadeh, Shadi; Johnson, Sindhu R.; Piotrowski, Pamela; Jewett, Lisa R.; Gordon, Jessica; Chung, Lorinda; Bilsker, Dan; Levis, Alexander W.; Turner, Kimberly A.; Cumin, Julie; Welling, Joep; Fortuné, Catherine; Leite, Catarina; Gottesman, Karen; Sauve, Maureen; Rodríguez-Reyna, Tatiana S.; Larche, Maggie; van Breda, Ward; Suarez-Almazor, Maria E.; Wurz, Amanda; Culos-Reed, Nicole; Malcarne, Vanessa L.; Mayes, Maureen D.; Boutron, Isabelle; Mouthon, Luc; Benedetti, Andrea; Thombs, Brett D.Abstract Background Systemic sclerosis (scleroderma; SSc) is a rare autoimmune connective tissue disease. We completed an initial feasibility trial of an online self-administered version of the Scleroderma Patient-centered Intervention Network Self-Management (SPIN-SELF) Program using the cohort multiple randomized controlled trial (RCT) design. Due to low intervention offer uptake, we will conduct a new feasibility trial with progression to full-scale trial, using a two-arm parallel, partially nested RCT design. The SPIN-SELF Program has also been revised to include facilitator-led videoconference group sessions in addition to online material. We will test the group-based intervention delivery format, then evaluate the effect of the SPIN-SELF Program on disease management self-efficacy (primary) and patient activation, social appearance anxiety, and functional health outcomes (secondary). Methods This study is a feasibility trial with progression to full-scale RCT, pending meeting pre-defined criteria, of the SPIN-SELF Program. Participants will be recruited from the ongoing SPIN Cohort ( http://www.spinsclero.com/en/cohort ) and via social media and partner patient organizations. Eligible participants must have SSc and low to moderate disease management self-efficacy (Self-Efficacy for Managing Chronic Disease (SEMCD) Scale score ≤ 7.0). Participants will be randomized (1:1 allocation) to the group-based SPIN-SELF Program or usual care for 3 months. The primary outcome in the full-scale trial will be disease management self-efficacy based on SEMCD Scale scores at 3 months post-randomization. Secondary outcomes include SEMCD scores 6 months post-randomization plus patient activation, social appearance anxiety, and functional health outcomes at 3 and 6 months post-randomization. We will include 40 participants to assess feasibility. At the end of the feasibility portion, stoppage criteria will be used to determine if the trial procedures or SPIN-SELF Program need important modifications, thereby requiring a re-set for the full-scale trial. Otherwise, the full-scale RCT will proceed, and outcome data from the feasibility portion will be utilized in the full-scale trial. In the full-scale RCT, 524 participants will be recruited. Discussion The SPIN-SELF Program may improve disease management self-efficacy, patient activation, social appearance anxiety, and functional health outcomes in people with SSc. SPIN works with partner patient organizations around the world to disseminate its programs free-of-charge. Trial registration ClinicalTrials.gov NCT04246528 . Registered on 27 January 2020