Dewi Guellec

ANCA-associated vasculitis in patients with primary Sjögren's syndrome: Detailed analysis of 7 new cases and systematic literature review

OBJECTIVES To describe the clinical presentation, management and prognosis of patients diagnosed with both primary Sjögrens syndrome (pSS) and anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV). METHODS French nation-wide survey completed by a systematic literature review. RESULTS This work identified 7 new cases of coexisting pSS and AAV: 2 microscopic polyangiitis (MPA), 2 granulomatosis with polyangiitis (GPA), 2 anti-myeloperoxidase (MPO)-ANCA renal-limited AAV, and 1 eosinophilic granulomatosis with polyangiitis (EGPA). The systematic literature search identified 15 previously published cases. Among the 22 patients, 19 were females. Mean age at diagnosis of AAV was 63.9±9.8years. All individuals with available information experienced at least one extra-glandular manifestation attributable to pSS. p-ANCA with anti-MPO specificity were found in 76.2% (16/21), c-ANCA with anti-PR3 specificity in 14.3% (3/21) and isolated c-ANCA in 13.6% (3/22). Vasculitis involved kidneys (n=13), lungs (n=8), skin (n=6), peripheral nerves (n=5), central nervous system (n=2), small bowel (n=1), muscle (n=1), ear chondritis (n=1) and sinuses (n=1). The mean AAV follow-up was 73.5 (±120.0) months. While on treatment, disease remission occurred in 77.3% of cases, and one death was reported in the first 6months after diagnosis. CONCLUSION This work shows that AAV may occur in patients with pSS. These are most commonly p-ANCA associated vasculitis with anti-MPO specificity. AAV may reveal an underlying pSS or arise during its evolution, but did not precede pSS in any of these cases. AAV occurrence appears to be correlated with extra-glandular manifestations of pSS.

Is there specific neurological disorders of primary Sjögren's syndrome?

Neurological manifestations of primary Sjögrens syndrome are multiple and appear frequently. Depending on data analysis, patient recruitment, and diagnosis criteria used to defined primary Sjögrens syndrome or neurological manifestations, the estimated prevalence is between 0 and 70%. Peripheral neurological complications seem the most common, particularly sensory-motors axonal neuropathies. Neuronopathy seems to be the most specific neurological complication of primary Sjögrens syndrome. Central manifestations of primary Sjögrens syndrome are not uncommon, but the neurological complications spectrum is not well defined. Neuromyelitis optica is regularly found among central complications. To conclude, although central and peripheral complications of primary Sjögrens syndrome are difficult to assess, partly because of the wide spectrum of possible manifestations, it is around 20%. Neuronopathy is still the most specific complication.

Relationship Between Hip Dysplasia, Pain, and Osteoarthritis in a Cohort of Patients with Hip Symptoms

Objective. The relationship between acetabular dysplasia (HD) and hip osteoarthritis (OA) remains unclear, especially for mild forms of dysplasia. Our objectives were to estimate the prevalence of HD in a population-based sample with symptoms and to evaluate potential associations linking HD, hip OA, and hip pain. Methods. Individuals 40 to 75 years of age with symptoms in 1 or both hips were recruited during a multiregional prevalence survey. All study participants underwent examination and radiographs. Radiographs were evaluated using Kellgren-Lawrence staging (with stages ≥ 2 indicating hip OA) and HD measures [center-edge (CE) angle, acetabular inclination angle (HTE), acetabular depth (AD), and vertical center-anterior margin angle]. Results. We studied both hips of 842 individuals (1684 hips), among whom 203 had hip OA. Compared to left hips, right hips had significantly smaller CE angles and significantly greater AD and HTE values (p ≤ 0.001). Overall, the prevalence of HD ranged from 7.6% to 22.2% of the hips depending on the measure used. The prevalence of HD was higher in individuals with hip OA, with significant differences for abnormal HTE (19.1% vs 11.4%; p < 0.0001) and abnormal CE (11.3% vs 7.5%; p = 0.04). By logistic regression, only abnormal HTE remained associated with OA. Same-side hip pain was not statistically more common in individuals with HD after stratification on OA status (p = 0.12). Conclusion. Our study confirms the relationship between OA and HD, particularly as defined based on the HTE angle.

Should non-steroidal anti-inflammatory drugs be used continuously in ankylosing spondylitis?

OBJECTIVE The 2010 update of ASAS/EULAR recommendations for managing ankylosing spondylitis (AS) specify that continuous non-steroidal anti-inflammatory drug (NSAID) treatment should be preferred in patients with persistently active, symptomatic disease. Here, our objective was to assess whether continuous NSAID therapy improves disease control and influences radiographic progression compared to on-demand therapy. We also assessed the safety profiles of both regimens. METHODS We performed a review by searching the PubMed and Embase databases using two MeSH term combinations to compare continuous and on-demand NSAID therapy in terms of disease control, radiographic progression, and safety. RESULTS The only study evaluating the impact of continuous NSAID therapy on disease control showed no significant difference with on-demand therapy. In four studies, continuous treatment was associated with slower radiographic progression, as assessed in three studies using the modified Stoke Ankylosing Spondylitis Spinal Score (m-SASSS). Three studies compared the safety of continuous and on-demand celecoxib, two in osteoarthritis and one in AS, and found no significant differences regarding the usual side effects of Cox-2 inhibitors. CONCLUSIONS Several studies showed slower radiographic progression with continuous NSAID therapy in AS. No studies demonstrated superiority of continuous NSAID therapy regarding symptom control. Continuous NSAID therapy (at least with Cox-2 inhibitors) does not modify safety compared to on-demand therapy.

Is routine ankle-brachial pressure index evaluation useful in rheumatoid arthritis?

Joint Bone Spine - In Press.Proof corrected by the author Available online since jeudi 26 juillet 2012

Eosinophilia predicts poor clinical outcomes in recent-onset arthritis: results from the ESPOIR cohort

Objectives To determine the prevalence of eosinophilia in patients with recent-onset arthritis suggestive of rheumatoid arthritis (RA) and to describe their features and outcomes. Methods We performed an ancillary study of data from a French prospective multicentre cohort study monitoring clinical, laboratory and radiographic data in patients with inflammatory arthritis of 6 weeks to 6 months duration. We determined the proportion of patients with eosinophilia, defined as a count >500/mm3, at baseline and after 3 years. Features of patients with and without baseline eosinophilia were compared. Results Baseline eosinophilia was evidenced in 26 of 804 (3.2%) patients; their mean eosinophil count was 637.7±107/mm3. Baseline eosinophilia was ascribed to atopic syndrome in 6 of 26 (23.1%) patients. After 3 years, patients with eosinophilia had higher Health Assessment Questionnaire scores (0.9 vs 0.5, p=0.004), higher patient visual analogue scale activity score and morning stiffness intensity (p=0.05), and were more often taking disease-modifying antirheumatic drugs (p=0.02). Baseline eosinophilia was not associated with presence of extra-articular manifestations. Conclusions Eosinophilia is rare in recent-onset arthritis suggestive of RA, and is usually directly related to the rheumatic disease. Our data suggest that patients with mild eosinophilia at diagnosis could respond worse to the treatment than those without.

Comparison of 2002 AECG and 2016 ACR/EULAR classification criteria and added value of salivary gland ultrasonography in a patient cohort with suspected primary Sjögren’s syndrome

BackgroundThe objective was to evaluate concordance between 2002 American-European Consensus Group (AECG) and 2016 American College of Rheumatology (ACR)/European League Against Rheumatism (EULAR) classification criteria for primary Sjögren’s syndrome (pSS) and to assess how salivary gland ultrasonography (SGUS) might improve the classification of patients.MethodsPatients with suspected pSS underwent a standardised evaluation, including SGUS, at inclusion into the single-centre Brittany DIApSS cohort. Agreement between the two criteria sets was assessed using Cohen’s κ coefficient. Characteristics of discordantly categorised patients were detailed.ResultsWe prospectively included 290 patients between 2006 and 2016, among whom 125 (43%) met ACR/EULAR criteria and 114 (39%) also met AECG criteria; thus, 11 (4%) patients fulfilled only ACR/EULAR, no patients AECG only, and 165 (57%) patients neither criteria set. Concordance was excellent (κ = 0.92). Compared to patients fulfilling both criteria sets, the 11 patients fulfilling only ACR/EULAR criteria had similar age and symptom duration but lower frequencies of xerophthalmia and xerostomia (p < 0.01 for each) and salivary gland dysfunction (p < 0.01); most had systemic involvement (91%), including three (27%) with no sicca symptoms; 91% had abnormal salivary gland biopsy and 46% anti-Sjögrens-syndrome-related antigen A (anti-SSA); 64% were diagnosed with pSS by the physician. SGUS was abnormal in 12% of the 165 patients fulfilling no criteria set. Including SGUS among the ACR/EULAR criteria increased sensitivity from 87.4% to 91.1% when physician diagnosis was the reference standard.ConclusionsAgreement between AECG and ACR/EULAR criteria sets is excellent. ACR/EULAR criteria are slightly more sensitive and classified some patients without sicca symptoms as having pSS. Including SGUS in the ACR/EULAR criteria may further improve their sensitivity.

Assessing polymyalgia rheumatica activity when C-reactive protein is unavailable or uninterpretable

Objective The PMR activity score (PMR-AS) includes the CRP value, which may be lacking or invalid owing to anti-IL-6 therapy. Our objective was to develop alternatives to PMR-AS that do not require CRP. Methods We used the Club Rhumatisme et Inflammation (CRI; 89 patients with PMR) and the Tolerance and Efficacy of tocilizumab iN pOlymyalgia Rheumatica (TENOR; 20 patients with recent-onset PMR naive to glucocorticoid who received three tocilizumab infusions, at weeks 0, 4 and 8, followed by prednisone from weeks 12 to 24) cohorts. In the CRI cohort, we evaluated correlations between PMR-AS items to select the best item for imputing CRP. Then we calculated the PMR-AS with (PMR-AS) and without (clin-PMR-AS) CRP and we used the linear regression between PMR-AS and clin-PMR-AS to obtain CRP-imputed (CRP-imp) PMR-AS. Finally, we evaluated agreement between clin-PMR-AS, CRP-imp PMR-AS, PMR-AS and ESR-PMR-AS in the TENOR cohort during tocilizumab therapy. Results In the CRI cohort, agreement between PMR-AS and clin-PMR-AS was excellent (κ = 0.90). Linear regression between PMR-AS and clin-PMR-AS [CRP-imp PMR-AS = 1.12(clin-PMR-AS)+0.26] allowed us to build the CRP-imp PMR-AS. Mean (s.d.) values were as follows: 8.40 (9.76) for PMR-AS, 7.24 (8.58) for clin-PMR-AS and 7.84 (9.61) for CRP-imp PMR-AS. CRP-imp PMR-AS agreed more closely with PMR-AS than did clin-PMR-AS. The results in the TENOR cohort confirmed that CRP-imp PMR-AS or ESR-PMR-AS could be used. Conclusion Alternatives to the PMR-AS obtained without CRP can be used to monitor PMR activity in everyday practice in patients without available CRP values and in those receiving IL-6 antagonist therapy.

Ultrasonography and magnetic resonance imaging changes in patients with polymyalgia rheumatica treated by tocilizumab

BackgroundThis study assessed inflammatory changes using ultrasound (US) and magnetic resonance imaging (MRI) in patients taking tocilizumab for polymyalgia rheumatica (PMR).MethodsEighteen patients were included in the prospective open-label TENOR study and received three tocilizumab infusions, without corticosteroids. B-mode and power Doppler US and MRI (T1 and T2-short time inversion recuperation weighted sequences) of the hips and shoulders were performed at weeks 0, 2, and 12. Subacromial, trochanteric, and iliopsoas bursitis and intraarticular glenohumeral and coxofemoral effusions/synovitis were scored from 0 to 3. Changes over time and US–MRI correlations were evaluated.ResultsAt baseline, the proportions of shoulders and hips with bursitis were 93 and 100% by MRI and 61 and 13% by US; and the corresponding proportions for intraarticular effusions/synovitis were 100 and 100% by MRI and 57 and 53% by US. Imaging findings did not improve during the first two treatment weeks. From baseline to week 12, bursitis improved significantly at all four joints by MRI (P = 0.005) and US (P = 0.029) and intraarticular effusions/synovitis by US only (P = 0.001). The proportion of abnormalities that improved by week 12 was 42% by MRI and 37% by US. MRI detected bursitis in a larger proportion of hips (73% versus 13%) and US in a larger proportion of shoulders (57% versus 28%), whereas no difference was found for intraarticular effusions/synovitis. At baseline, agreement between US and MRI findings was poor.ConclusionsUS and MRI showed significant improvements in inflammatory lesions during tocilizumab treatment of PMR.

Evolution of early arthritis to spondyloarthritis after a 10-year follow-up.

Joint Bone Spine - In Press.Proof corrected by the author Available online since mardi 11 decembre 2012