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ID PMID Title PublicationDate abstract
20621335 Tocilizumab in refractory adult-onset Still's disease with aseptic meningitis--efficacy of 2011 Feb Adult-onset Still's disease is a multisystem inflammatory disorder of unknown etiology characterized by typical spiking fever, evanescent rash, arthralgia, and leucocytosis. Neurologic manifestations are infrequent, seen in 7 to 12% of cases. We present the case of a young male admitted with aseptic meningitis that satisfied the diagnostic criteria of Adult-onset Still's disease. Refractoriness to therapy with corticosteroids and cyclosporine A led to the use of humanized monoclonal anti-interleukin-6 receptor antibody "tocilizumab" with dramatic response. The case is reported for the rarity of presentation and the need to consider the diagnosis in related clinical scenarios. Also, current literature on the use of tocilizumab in intractable disease is reviewed.
22516992 [Analysis of the clinical features at onset of primary Sjögren's syndrome]. 2012 Apr 18 OBJECTIVE: To comprehend clinical features at onset of primary Sjogren's syndrome (pSS) in order to provide useful data for its clinical management. METHODS: In the study, 224 patients diagnosed with pSS in the Department of Rheumatology and Immunology of Peking University People's Hospital from Jun. 1st, 2007 to Aug. 1st, 2008 were investigated, including gender, age of onset, time and site of first hospitalization and definite diagnosis, etc. RESULTS: In this 224 pSS cohort (213 females and 11 males), the male/female ratio was 1:19.4, the mean age of onset was (53.5±11.7) years, and median duration was 9.4 years (ranging from 0.2 to 40.0 years).The manifestations showed that up to 33% of the patients (74/224) had leukopenia, 25% (56/224) polyarthralgia, 16.5% (37/224) raynaud phenomenon, 15.6% (35/224) hepatic injury, 12.1% (27/224) pulmonary interstitial fibrosis, 11.6% (26/224) purpuras on lower extremities, 8.0% (18/224) hemogram abnormal, 5.8% (13/224) thrombopenia, and 3.6% (8/224) renal tubule acidosis. When the risk factor of the systemic involvements, was analyzed, two factors were significantly associated with pulmonary interstitial fibrosis: age (OR=1.074, 95% CI=1.031-1.118), and duration (OR=1.075, 95% CI=1.023-1.128). Liver involvement was associated with duration (OR=1.050, 95% CI=1.002-1.100). In addition, 8.0% of the pSS patients(18/224)showed family history of autoimmune diseases and 11.2%(25/224)had family history of tumor. CONCLUSION: In this cohort of the pSS patients, female is predominant and the incidence of extro-glandular manifestations, such as leukopenia, lung and liver involvements is high, and pSS has inheritance intention.
22226370 IL-21 and Sjögren's syndrome. 2011 Treatment of Sjögren's syndrome is almost entirely symptomatic. A lack of true understanding of the underlying immunological pathology of the disease prevents directed therapy. Interleukin-21 (IL-21) is elevated in the serum of patients with this disease and is expressed by the lymphocytes infiltrating the salivary glands. The known functions of IL-21 in facilitating differentiation, proliferation, and survival of both B and T cells mesh well with the findings in Sjögren's syndrome. Demonstration of IL-21 as a fundamental aspect of the pathophysiology of Sjögren's syndrome could lead to the development of anti-IL-21 therapy for this disease.
22669597 Fluorodeoxyglucose (FDG) uptake in pulmonary rheumatoid nodules diagnosed by video-assiste 2013 Mar Two cases of rheumatoid nodules evaluated by fluorodeoxyglucose (FDG) positron emission tomography/computed tomography (PET/CT) and video-assisted thoracic surgery (VATS) biopsy are reported. The first case was that of a 44-year-old woman who presented with a cavitated nodule with intense standardized uptake values (SUVs) both in the early (max 3.4) and delayed (max 4.4) phases, suggesting malignancy. However, after VATS biopsy, she was diagnosed as having a rheumatoid nodule with vasculitis. The second case was that of a 74-year-old woman admitted with bilateral lung nodules, two of which showed intense early (max 2.2) and delayed (max 6.0) phase SUVs, and mild early (max 0.6) and delayed (max 0.9) phase SUVs. These two nodules were finally proven to be a lung cancer and rheumatoid nodule without vasculitis, respectively. These cases show that rheumatoid nodules with an enhanced inflammatory process, such as vasculitis, can appear false-positive for malignancy on FDG-PET/CT scan images.
22655584 [Adult refractory Still disease with atypical articular manifestations: efficacity of inte 2012 May INTRODUCTION: Tocilizumab is a humanized monoclonal antibody directed against interleukin-6 receptor and is beginning to be reported as effective in some cases of Still's disease refractory in adults (ASD). ASD is rare, heterogeneous, with unpredictable evolution. The distal destructive arthritis represents a possible complication. PATIENT: We report an unusual case of adult-onset Still's disease with severe distal interphalangeal destructive arthritis with refractory early and prolonged remission after the first tocilizumab infusion. CONCLUSION: Tocilizumab can be used in patients with refractory ASD after failure or intolerance of conventional treatments.
22353900 [Clinical assessment of oral diagnostic items in 2002 classification criteria for primary 2012 Feb 18 OBJECTIVE: To evaluate the diagnostic value of each item in 2002 international classification criteria for primary Sjogren's syndrome (pSS) in clinically diagnosed cases. METHODS: All patients were from the Department of Oral Medicine of Peking University School of Stomatology from 2005 to 2010. Their clinical manifestations and lab testing results met the standard criteria and the diagnosis was established according to international classification criteria (2002). We retrospectively collected all of the clinical and information and did the data analysis. RESULTS: A total of 148 pSS patients were included in the study. When the oral evaluations were performed, 98.0% of the patients complained dry mouth, 96.6% of them had decreased non-stimulated salivary flow rate, and 60.1% had positive parotid sialography results, which was consistent with diagnosis. It was found out in the lab testing that 79.7% of the patients had positive anti-SSA antibody, and 75.0% of them had elevated globulin level. 20.3% of the patients were finally diagnosed by performing the lower lip biopsy. CONCLUSION: When applying 2002 international classification criteria for the diagnosis of pSS patients, oral symptoms and unstimulated salivary flow rate are feasible and sensitive, which play an important and indicative role in the diagnosis of pSS.
21591859 Two-year outcome of partial lacrimal punctal occlusion in the management of dry eye relate 2011 Jun PURPOSE: To analyze the influence of thermal partial punctal occlusion on the ocular surface of dry eye related to Sjögren syndrome. MATERIAL AND METHODS: Thirty-seven eyes of 19 patients (3 male and 16 female; 49.11 ± 14.33 years old) with keratoconjunctivitis sicca were enrolled in this study. Superior and inferior partial occlusion were performed in both eyes under topical anesthesia using thermal cautery with a sterile tip to obtain lacrimal punctum smaller than 0.5 mm. Schirmer I, break-up-time, diameter of lacrimal puncta, corneal fluorescein, and rose Bengal staining scores were analyzed before and after 24 weeks and after 24 months of the procedure. All measurements were performed under controlled climate. RESULTS: The average lacrimal punctum diameter before the procedure was 0.65 ± 0.134 mm. All lacrimal puncta were successfully reduced to less than 0.5 mm after 4 weeks of the procedure. The average Schirmer I test values improved statistically after 24 weeks and maintained stable after 24 months. Average break-up-time, rose Bengal, and fluorescein staining score values improved statistically after 24 weeks and improved even more after 24 months. Average Schirmer I test, break-up-time, rose Bengal, and fluorescein staining scores showed significant improvement (p < 0.0001) after 24 months of partial thermal punctal occlusion. CONCLUSION: Our study showed that reducing the punctum diameter to 0.5 mm can improve vital staining scores, break-up-time, and Schirmer I test in dry eye related to Sjögren syndrome.
22696669 Periodontal infection in adult-onset Still's disease patient: clinical and haematological 2011 Apr 26 In this case report, the authors described the first case of a patient with adult-onset Still's disease (AOSD) who presents advanced periodontal infection. AOSD is a rare systemic inflammatory disorder of unknown aetiology, characterised by spiking fever, usually exceeding 39°C, an evanescent salmon pink rash, arthritis and multiorgan involvement. Periodontal infection is a pathogen-induced oral inflammatory disease affecting the supporting tissues of teeth and is currently considered as a risk factor for cardiovascular disease. Several cytokines capable of inducing systemic effects are produced during the course of this infection and the values of serum markers of inflammation, such as C reactive protein (CRP), may significantly decrease after periodontal treatment. Although AOSD can produce elevations in CRP, similar increase may be produced by periodontal infection, suggesting the need for medical and dental diagnosis when evaluating the sources of acute-phase responses in systemic autoimmune disease patients.
20921900 Macrophage activation syndrome: why and what should a gastroenterologist know. 2011 Mar We recently treated a patient with adult-onset Still's disease who developed macrophage activation syndrome (MAS) secondary to disseminated histoplasmosis while being treated with adalimumab. The gastroenterology service was consulted early, before diagnosis, as the patient presented with elevated liver enzymes and disseminated intravascular coagulation. MAS is an exaggerated immune response that can develop as a primary condition or secondary to infections, drugs and various diseases, resulting in liver dysfunction, encephalopathy, pancytopenia and disseminated intravascular coagulation. The development of MAS has also been reported in patients with inflammatory bowel disease and post-liver transplantation and has been triggered by medications used by gastroenterologists, particularly sulfasalazine and anti-tumor necrosis factor biologic modifiers. Therefore, we present a review on etiology, pathogenesis, clinical and laboratory features, and treatment of MAS with a focus on gastrointestinal aspects and presentations. MAS is a life threatening condition with a high mortality rate if untreated. Therefore it is important to recognize this condition early. As these patients may occasionally present to gastroenterologists we hope this review will increase awareness of this rare, but serious syndrome.
22580050 Still's disease and the mitochondrion: the other face of an old friend? 2012 Aug Although Still's disease has been first described more than one century ago, it still appears as an orphan entity, which should be separated from the other auto-inflammatory diseases (AID). The main reason to individualize Still's disease among the AID is the absence of any genetic predisposition. Recently, the human mitochondria have been clearly implicated in the systemic inflammation that is observed during the innate immune response. After various types of cellular injuries, including infections, the release of "Damage-Associated Molecular Patterns" (DAMPs) from mitochondria can recruit circulating polymorphonuclear neutrophils (PMNs), monocytes and macrophages, along with the activation of NLRP3 inflammasome. Flares of Still's disease usually mimic systemic bacterial infections, with high levels of PMNs, but no evidence of circulating bacteria. Ubiquitous and usually benign viruses, such as human herpes virus 6 (HHV-6), appear capable of inducing mitochondrial damages. Such a phenomenon could in turn initiate the flares of Still's disease, thereafter persisting as an inappropriate and self-perpetuating reaction to an endogenous bacterial vestige, the mitochondrion itself.
21946464 Clarithromycin in adult-onset still's disease: a potentially useful therapeutic. 2011 Oct Adult-onset Still's disease (AOSD), an autoinflammatory syndrome of unknown etiology, typically manifests with spiking fevers, polyarthritis, and characteristic evanescent rash. We describe a young woman with AOSD complicated by calf fasciitis that serendipitously responded to clarithromycin administered for another indication. Remarkable improvement followed rechallenges with clarithromycin for subsequent AOSD flares. In addition to their antibacterial actions, macrolides demonstrate immunomodulatory effects, including suppression of proinflammatory cytokine production and neutrophil action. Previous clinical trials provide promising preliminary evidence of a therapeutic effect of macrolides in chronic inflammatory diseases. Although AOSD pathogenesis remains unclear, a role for dysregulation of innate immunity is supported by recent literature. Based on this possible innate immune mechanism, we suspect that macrolides may have induced a therapeutic response in this patient with AOSD. A clinical trial is warranted to establish or refute their therapeutic efficacy.
21853681 [Still's disease or systemic lupus erythematosus in a young adult patient--diagnostic doub 2011 Systemic lupus erythematosus and Still's disease are chronic autoimmune disorders of unknown etiology. Symptomatology of these diseases may be similar causing diagnostic difficulties. Long-term observation and immunological studies are essential to identify the definite disorder. We present a case of a 24-year-old patient with high fever, sore throat and arthritis. During hospitalization rash accompanying fever, nodular erythema, pulmonary changes, liver damage and splenomegaly were observed. Although initially adult-onset Still's disease was diagnosed according to the Yamaguchi criteria, the diagnosis of systemic lupus erythematosus was made after re-analysis of the clinical course and immunological tests.
21364049 Patients considered as having undifferentiated peripheral inflammatory arthritis: a system 2011 Mar OBJECTIVE: To systematically review the differential diagnosis and minimal clinical investigation used prior to making a diagnosis of undifferentiated peripheral inflammatory arthritis (UPIA). METHODS: A systematic literature search was performed for articles published between January 1950 and December 2008 in Medline and Embase, and for abstracts presented at the 2007 and 2008 meetings of the American College of Rheumatology (ACR) and European League Against Rheumatism (EULAR). Studies including defined cohorts of patients with UPIA were retrieved according to predefined inclusion/exclusion criteria. Selected studies were systematically reviewed and relevant data extracted. Baseline characteristics were also recorded to obtain a clinical picture of patients classified as UPIA. RESULTS: Seventy-four articles were included. Of those, 52 reported baseline characteristics. Tremendous variation existed among studies, reflecting the different inclusion/exclusion criteria used. Rheumatoid arthritis, spondyloarthropathies, osteoarthritis, crystal arthritis, connective tissue diseases, and infections were the most common diagnoses of exclusion for UPIA and made up the other subsets of patients in cohorts with mixed populations. The baseline investigation undertaken prior to diagnosis of UPIA was reported in 7 articles. History, physical examination, tender and swollen joint count, rheumatoid factor, HLA-B27, erythrocyte sedimentation rate, C-reactive protein, and radiographs of hands and feet were the only items mentioned in at least 50% of the reports. CONCLUSION: Studies of UPIA are heterogeneous. Few studies reported on the minimal clinical investigation necessary to arrive at a diagnosis of UPIA. Differential diagnosis usually consisted of the most common rheumatologic conditions but could be vast.
23097701 Usefulness of patients-reported outcomes in rheumatoid arthritis focus group. 2012 Objective. Patient-reported outcomes (PROs) have become an essential part of the assessment of patients with rheumatoid arthritis (RA). We aimed to evaluate the agreement and correlation between PROs and the physician's measurements. Methods. This was a cross-sectional analytical study in which 135 patients with RA were clinically evaluated during two different sessions of focus group interviews. Rheumatologist recorded 28 swollen (SJCs) and tender joint counts (TJCs). The patients filled out the PROs instruments (MDHAQ, RADAI, RAPID3, 4, and 5 and self-report articular index (SAI) diagram for pain and joint swelling). DAS28 was calculated (C-reactive protein). An adjusted multiple lineal regression model was done (DAS28 as dependent variable). Results. Highly significant agreements were found between SJC and TJC registered by the physician and patient. There was moderate correlation between DAS28 with patient SJC (r = 0.52), patient TJC (r = 0.55), RADAI (r = 0.56), RAPID3 (r = 0.52), RAPID4 (r = 0.56), RAPID5 (r = 0.66), and VAS-Global (r = 0.51). Likewise, we found moderate to high correlations between CDAI and SDAI with all variable measurements done by the patients. The resulting predictive equation was DAS28(CRP) = 2.02 + 0.037 × RAPID4 + 0.042× patient SJC. Conclusion. PROs applied in focus groups interview are a useful tool for managing patients with RA regardless of gender, educational level, and duration of disease.
23035013 Utility of the new rheumatoid arthritis 2010 ACR/EULAR classification criteria in routine 2012 OBJECTIVES: The new 2010 American College of Rheumatology/European League Against Rheumatism (ACR/EULAR) criteria for rheumatoid arthritis (RA) have been designed to classify early onset RA, but has not been studied to identify RA in patients with arthritis seen in routine clinical care where correct 'classification' of patients, when they are not selected for having RA would be important. DESIGN: Prospective, consecutive patients cohort. SETTING: Outpatient clinic of a university rheumatology centre. PARTICIPANTS: A total of 126 patients with joint symptoms were consecutively recruited. INTERVENTIONS: The ACR/EULAR RA criteria were applied, with questions followed by a targeted musculoskeletal exam. The gold standard for the diagnosis of RA was the primary rheumatologist's diagnosis. PRIMARY OUTCOME MEASURE: Number of patients with non-RA diagnosis who were classified as having RA by the new classification criteria. RESULTS: The sensitivity and specificity of the 2010 criteria in classifying RA were 97% and 55%, respectively, compared with the 1987 RA criteria which were 93% and 76%, respectively. The 2010 criteria as applied to this group of patients had a poorer positive predictive (44% vs 61%) and a similar negative predictive value (98% vs 97%) compared with the 1987 criteria. More specifically, 66.7% of systemic lupus erythematosus patients, 50% of osteoarthritis, 37.5% of psoriatic arthritis and 27.2% of others fulfilled the new criteria and could have been classified as RA. CONCLUSIONS: In this, we believe, the first study to examine the new 2010 ACR/EULAR RA criteria among consecutive patients seen in routine care, we found the criteria to have low specificity, and therefore incorrectly label those as having RA when, in fact, they may have a different type of inflammatory arthritis. Physicians need to be aware of this when applying the new criteria for classifying their patients for any purpose.
22013422 Current medical treatments for juvenile idiopathic arthritis. 2011 Juvenile idiopathic arthritis (JIA) differs markedly from adult rheumatoid arthritis. It is not a single disease, but an exclusion diagnosis that gather together all forms of arthritis that begin before the age of 16 years, persist for more than 6 weeks, and are of unknown origin. The advent of the new biological treatments has dramatically changed both the observed responses to treatment and the expectations of therapies. The implementation of an adequate legislation as well as the presence of international research networks of pediatric rheumatology have contributed to foster the conduct of controlled clinical trials and the development of validated outcome measures. This review will currently describe the methodological approach for performing clinical trials in JIA as well as the current available drug treatment.
20858143 Hereditary isolated metatarsophalangeal arthritis. 2011 Jan OBJECTIVE: To describe a family with 13 members in four generations affected by early-onset isolated painful arthritis limited to the first metatarsophalangeal (MTP) joint but without evidence of generalized joint disease at follow-up. METHODS: A complete family pedigree was constructed and radiographs from the affected family members and their offspring were taken. Laboratory tests including serum measurements of C-reactive protein (CRP), urate, and rheumatoid factor (RF) were performed to exclude gout and rheumatoid arthritis from the diagnosis. RESULTS: The age at onset of first MTP joint symptoms varied from 12 to 51 years. Both females and males were affected in the four successive generations, including male-to-male transmission as well as maternal inheritance. The affected patients were often treated surgically with good pain-relieving results. CONCLUSION: To our knowledge, this is the first report of early-onset isolated foot metatarsal arthritis with apparent autosomal dominant inheritance.
22778567 Assessment of collagen-induced arthritis using cyanine 5.5 conjugated with hydrophobically 2012 Jul OBJECTIVE: To evaluate the potential and correlation between near-infrared fluorescence (NIRF) imaging using cyanine 5.5 conjugated with hydrophobically modified glycol chitosan nanoparticles (HGC-Cy5.5) and (18)F-fluorodeoxyglucose-positron emission tomography ((18)F-FDG-PET) imaging of collagen-induced arthritis (CIA). MATERIALS AND METHODS: We used 10 CIA and 3 normal mice. Nine days after the injecting collagen twice, microPET imaging was performed 40 minutes after the intravenous injection of 9.3 MBq (18)F-FDG in 200 µL PBS. One day later, NIRF imaging was performed two hours after the intravenous injection of HGC-cy5.5 (5 mg/kg). We assessed the correlation between these two modalities in the knees and ankles of CIA mice. RESULTS: The mean standardized uptake values of (18)F-FDG for knees and ankles were 1.68 ± 0.76 and 0.79 ± 0.71, respectively, for CIA mice; and 0.57 ± 0.17 and 0.54 ± 0.20 respectively for control mice. From the NIRF images, the total photon counts per 30 mm(2) for knees and ankles were 2.32 ± 1.54 × 10(5) and 2.75 ± 1.51 × 10(5), respectively, for CIA mice, and 1.22 ± 0.27 × 10(5) and 0.88 ± 0.24 × 10(5), respectively, for control mice. These two modalities showed a moderate correlation for knees (r = 0.604, p = 0.005) and ankles (r = 0.464, p = 0.039). Moreover, both HGC-Cy5.5 (p = 0.002) and (18)F-FDG-PET (p = 0.005) imaging also showed statistically significant differences between CIA and normal mice. CONCLUSION: NIRF imaging using HGC-Cy5.5 was moderately correlated with (18)F-FDG-PET imaging in the CIA model. As such, HGC-Cy5.5 imaging can be used for the early detection of rheumatoid arthritis.
23078992 Psoriatic arthritis: treat-to-target. 2012 Jul Recently, the concept of 'treat-to-target' has emerged as a topic of great interest in rheumatology, particularly as regards the therapeutic approach to patients with rheumatoid arthritis RA). From observational data as well data from controlled clinical trials, there is a body of evidence supporting this idea. Thus, closely monitoring RA patients and adjusting therapies with the goal of achieving the lowest disease activity possible can result in optimal outcomes for patients. Based on the success in RA, interest in adopting a treat-to-target approach in other rheumatic conditions, including psoriatic arthritis (PsA) has arisen. It would appear logical that some data from 'treat-to-target' approaches in RA may readily be extrapolated to PsA, particularly as it relates to PsA patients with polyarticular peripheral arthritis. However, PsA is a heterogeneous disorder, with involvement in areas quite distinct from RA, including the skin and nails, the axial spine, and the entheses. Therefore, developing a treat-to-target strategy in PsA will require additional disease specific considerations to optimise its implementation.
21362787 Inflammatory musculoskeletal disease: identification and assessment. 2011 Mar Diagnosis of psoriatic arthritis (PsA) is complex because not all patients with psoriasis and musculoskeletal symptoms of pain, stiffness, and dysfunction have PsA. Instead, they may have other inflammatory conditions such as rheumatoid arthritis, gout, or septic arthritis, or noninflammatory conditions such as osteoarthritis, recurrent tendonitis, mechanical back pain, or a myriad other musculoskeletal conditions. To acquire skill in diagnosing and monitoring the disease course of PsA, a clinician must recognize that there are multiple clinical domains that may be affected, including peripheral joints, entheseal insertion sites, dactylitis, and the spine. They must also appreciate the clinical features (history and physical examination) that are characteristic of immunologic inflammation and know how to utilize and interpret laboratory and imaging studies. Rheumatologists are expected to be skilled in these assessments. It is also helpful for dermatologists, primary care physicians, and other clinicians who work with psoriasis patients to have a working knowledge of assessments in PsA in order to identify and triage the patient for optimal management. Features that assist identification and assessment of PsA are reviewed in this article.