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ID PMID Title PublicationDate abstract
7675258 [Tracheal intubation in a case of Still's disease]. 1995 Mar The authors report the case of a patient with juvenile rheumatoid arthritis (Still's disease) who underwent laparotomic hysterectomy under general anesthesia. Tracheal intubation was performed with the aid of a fibroscope, as recommended in the literature, given that impairment of the cervical spine and the temporo-mandibular joint made it extremely unlikely that direct laryngoscopy could be performed. The authors also report the methods recommended by other authors in order to guarantee a sufficiently deep anesthetic plane together with patency of the airways in patients suffering from Still's disease.
8869216 Adult Still's disease: part I. Manifestations and complications in sixty-five cases in Fra 1995 Dec DESIGN: a retrospective multicenter study conducted in France identified 65 cases of adult Still's disease. Data were recorded on a standardized questionnaire validated by the Inflammatory Joint Disease and Immunorheumatology Committee of the Société Française de Rhumatologie. OBJECTIVES: (1) To compare clinical and laboratory findings in our patients with those reported in earlier studies, particularly two large series from Canada and Japan, respectively; (2) to describe the systemic and visceral complications associated with adult Still's disease. RESULTS: as compared with the two above-mentioned series, our study group included more patients who had experienced onset of their disease after the age of 35 years and fewer patients with involvement of the liver, spleen, or lymph nodes. Rates of occurrence of arthritis, myalgia, sore throat, pleuritis, pericarditis, and abdominal pain were significantly higher in the Canadian series than in the other two series. Arthritis was absent in one fourth of our patients. Life-threatening complications included "Still's hepatopathy", disseminated intravascular coagulation (with hemophagocytosis in some cases), and "Still's myocarditis". CONCLUSION: differences in the expression of adult Still's disease were found between patients from Canada, France, and Japan. Adult Still's disease can be responsible for life-threatening complications.
7569721 Evaluation of the immune status in patients with primary and secondary Sjögren's syndrome 1995 The total white blood cell count, percentage and total count of lymphocytes, CD3+, CD4+, and CD8+ subpopulations of T-lymphocytes, population of CD20+ B-lymphocytes, the ability of polymorphonuclears to phagocyte yeasts, the ability of polymorphonuclear cells to kill serum-resistant strain of E. coli and O2-dependent killing capacity of polymorphonuclears were evaluated in a group of 10 patients with the sicca syndrome and in a group of 9 patients with secondary Sjögren's syndrome and in a healthy control group. There was found a significant decrease in the relative and absolute counts of CD4+ helper/inducer T cells and in the activity of phygocytic cells in both groups of patients with Sjögren's syndrome as compared with the control group. There was no significant difference in the evaluated parameters between primary and secondary Sjögren's syndrome patients.
8092630 [Systemic signs of primary Gougerot-Sjögren syndrome. 48 cases]. 1994 Forty-eight patients with primary Sjögren syndrome are documented together with the results of their baseline investigations. The majority of patients were female (44 out of 48) and mean age was 63.2 years. Common clinical features included 20 parenchymal lung disease, among which 4 had interstitial pulmonary fibrosis and 2 lymphocytic interstitial pneumonitis, 12 neurologic manifestations, 16 Raynaud's phenomenon, 12 arthritis, and 3 gastrointestinal involvement. Haematological features occurred in 15 patients and another autoimmune disease was encountered in 13 cases. These extraglandular manifestations were the dominating reasons for hospital referral in 43.5% of cases, the sicca syndrome were most often only mentioned by the patients after special questioning which explain considerable delay before the diagnosis.
8463619 Primary Sjögren's syndrome associated with chronic atrophic gastritis and pernicious anem 1993 Mar Sjögren's syndrome (SS) is a systemic, autoimmune exocrinopathy in which destructive, inflammatory-cell infiltration can affect any gland. Although chronic atrophic gastritis is the most common form of gastrointestinal involvement in SS, pernicious anemia develops in rare cases. We therefore report a patient with these three associated diseases.
8729607 Dyspareunia in undiagnosed Sjogren's syndrome. 1996 Jan A case of superficial dyspareunia in a 46-year-old women is described. Clinical suspicion, examination and investigations supported a diagnosis of Sjogren's syndrome.
7497800 Usual interstitial pneumonia associated with primary Sjögren's syndrome. 1995 Dec We describe the first case, to our knowledge, of usual interstitial pneumonia (UIP) as the pulmonary manifestation in primary Sjögren's syndrome (SjS). A 45-year-old woman was admitted to our hospital because of a dry cough and an interstitial shadow on a chest roentgenogram. Labial biopsy and sialogram confirmed a diagnosis of SjS. BAL fluid analysis revealed lymphocytosis with a decreased CD4/CD8 ratio compatible with bronchiolitis obliterans organizing pneumonia or lymphoid interstitial pneumonia. Open-lung biopsy specimen, however, showed evidence of UIP. Open-lung biopsy was a useful and necessary examination to determine the nature of the pulmonary complication in primary SjS. Conservative treatment without corticosteroids maintained a stable condition for a follow-up period of 3 years.
8583730 [Primary Sjögren's syndrome accompanied by pleural effusion]. 1995 Nov A 40-year-old woman was admitted to the hospital because of fever and right cervical pain. Facial erythema developed after admission, along with interstitial shadows and pleural effusion in both lower lung fields. Microscopical examination of transbronchial lung biopsy specimens showed infiltration by lymphocytes and thickening of alveolar walls. Analysis of bronchoalveolar lavage fluid revealed an abnormally high number of cells and CD8 lymphocytes. Schirmer's test and a gum test showed abnormally low lacrimation and salivation. Anti-nuclear antibody, and antibodies against SS-R, and SS-B were present. These findings led to the diagnosis of primary Sjögren's syndrome accompanied by pleural effusion. Facial erythema was regarded as a symptom of Sjögren's syndrome. Prednisolone was given (60 m/day), and the symptoms and the abnormality on the chest roentgenogram resolved within about 1 month.
7774460 [A histopathologic study on lacrimal gland in Sjögren's syndrome]. 1994 Nov Biopsy specimens of palpebral lacrimal glands from 15 cases of Sjögren's syndrome (SS) were studied by light and transmission electron microscopy. Six cases without any symptoms or signs of dry eyes were used as the control. The main changes of SS included: (1) massive infiltration of lymphocytes; (2) atrophy of lacrimal glandular follicles and ducts in various degrees; (3) degeneration and necrosis of glandular epithelium; and (4) proliferation of connective tissue. Lymphatic infiltration indicates that the damage of the glandular tissue might be related to autoimmune pathogenesis. According to the histopathologic findings, the authors divide SS into three stages which are basically consistent with their clinical presentations.
1450803 Injection therapy of superficial rheumatoid nodules. 1992 Nov Although intralesional corticosteroid injection of subcutaneous rheumatoid nodules was mentioned in 1968, this simple procedure is not commonly practised. A placebo-controlled, double-blind trial of intralesional corticosteroid injection using 24 rheumatoid nodules from 11 patients was carried out to determine the efficacy and safety of the procedure. Nodules injected with methylprednisolone and lignocaine regressed significantly more than nodules injected with placebo (lignocaine alone). This was consistently shown in all modalities of assessments which included patients' assessments (P < 0.001) and investigator's assessments (P < 0.001) of the percentage change in nodule size, and gross measurements of nodule volumes using a pincer (P < 0.001). Nine of 12 active injections produced > or = 50% loss in nodular volume with complete disappearance of two nodules. This compares with only one out of 12 placebo injections which resulted in > or = 50% loss in nodular volume. The patients found all 12 active injections to be worthwhile compared to only two of 12 placebo injections being worthwhile. The only complication of injection therapy observed was that of pain during the procedure.
1586249 Beta 2 microglobulin in tear fluid from patients with primary Sjögren's syndrome. 1992 Apr beta 2 Microglobulin concentration in tear fluid was measured in 35 patients with primary Sjögren's syndrome (SS), in 28 normal control subjects matched for age and sex, and in 18 patients with arthralgias or myalgias and sicca complaints, in whom the diagnosis primary SS had been excluded. Increased beta 2 microglobulin concentrations were found in the patients with SS, but no correlation was found with the duration of the disease, age, or tear fluid production. Tear fluid beta 2 microglobulin determinations may be useful as an adjunctive diagnostic test for primary SS.
8643895 [A 17-year-old female patient with recurring fever, chills, exanthema, myalgia and polyser 1996 Mar 12 The 17-year-old was admitted for investigation of a fever persisting for three weeks in spite of antibiotic treatment. Based on the clinical picture presenting with fugitive exanthema during febrile episodes, myalgia, polyserositis, leucocytosis with toxic granulations and-- after an antibiotic window--negative cultures of all investigated fluids (blood, pleural and peritoneal fluid), adult-type Still's disease was diagnosed. Treatment with steroids and indomethacine was only temporarily successful. Therapeutic stabilization first occurred under administration of phenylbutazone. The course was complicated by three surgical abdominal interventions because of an unclear acute abdomen, a strangulation ileus and a small-bowel perforation.
7553056 [The first case of Sjögren's syndrome with anti-Wa antibody]. 1995 Apr A 42-year-old woman presented in 1992 with Raynaud's phenomenon, polyarthralgia and morning stiffness and next year developed xerostomia and xerophthalmia. Investigations revealed 20 mm/hr of ESR, positive test for RAPA (1 : 320), ANF (1 : 1280), anti-SS-A antibody and anti-Wa antibody, and positive Schirmer tear test. A biopsied specimen of minor salivary gland of lip showed lymphocytic infiltration around the ducts and fibrotic changes and a sialogram demonstrated diffuse dilatations of the peripheral ducts. Resulting from the data shown above, the diagnosis of Sjögren's syndrome was made. Anti-Wa antibody, which recognizes a 48 kD tRNA associated protein was identified in serum from a patient with systemic sclerosis by Yamagata in 1985. So far the antibody has been specific for systemic sclerosis, because all of six patients with anti-Wa antibody previously reported were diagnosed as having systemic sclerosis. However, we here described the first case of Sjögren's syndrome with anti-Wa antibody.
8052929 [Cyclophosphamide-responsive subclinical Sjögren's syndrome in a patient with initial per 1994 Jun A 36-year old woman was admitted because of painful dysesthesia of her extremities, suggesting the presence of mononeuritis multiplex. Laboratory data was almost within normal limits, with the exception of lupus anticoagulant positivity and increase of IgM level. We considered the possibility of connective tissue diseases and examined the patient accordingly. Keratoconjunctivitis sicca without dry eye symptoms, identified by rose-bengal and fluorescence testing, was the only recognizable abnormality. Oral sicca symptoms were not revealed although lip biopsy showed infiltration by a moderate number of plasma cells and lymphocytes. Under the diagnosis of subclinical Sjögren's syndrome, the following examination was carried out. Sural nerve biopsy specimens revealed wallerian degeneration and perivascular mononuclear cell infiltration of the vasa nervorum. We therefore concluded that the peripheral neuropathy was caused by subclinical Sjögren's syndrome. Magnetic resonance imaging (MRI) of the brain demonstrated multiple small lesions with increased spin echo images (T2 weighted) in the white matter. So, this patient was suffered from not only peripheral but also central nervous system complications. The mechanism of nervous system involvement was considered to be mononuclear cell-dependent ischemic damage caused by infiltration of the vasa nervorum. Both steroid pulse therapy and oral corticosteroid administration were ineffective in treatment of the peripheral neuropathy. Alternative use of cyclophosphamide (75 mg per day) was dramatically effective in relieving peripheral nervous system disorders. This was evident in the remarkable improvement of painful dysesthesia, grip strength and motor nerve conduction velocities. This case could be considered valuable for understanding the pathophysiology of Sjögren's syndrome and associated nervous system complications.
8258233 Childhood systemic lupus erythematosus in songklanagarind hospital: a potential unique sub 1993 Sep Twenty systemic lupus erythematosus patients under the age of 16 who had attended the Pediatric Department of Songklanagarind Hospital in the period 1985-1991 were reviewed retrospectively. The most common complaints were fever, alopecia, malar rash and oedema. The girl/boy ratio was 2.3/1. There were many unusual presentations such as Stevens-Johnson syndrome, psoriasis, rheumatoid nodules, papillitis and pseudotumor cerebri. Nephritis, haematological disorder and CNS involvement were found in 80, 50 and 35% respectively. All cases were positive for ANA. Sixty-five percent of cases had complications and mostly more than one. Common complications were infection, renal complication and side effects of chemotherapy. Five patients died because of renal failure, severe infections, cerebral infarction or GI bleeding.
8235664 Comparative efficacy and safety of advanced drug therapy in children with juvenile rheumat 1993 Aug Results from three randomized placebo-controlled trials were combined in a meta-analysis to compare the clinical utility of four advanced drug therapy agents used to treat juvenile rheumatoid arthritis (JRA): D-penicillamine (10 mg/kg/d), hydroxychloroquine (6 mg/kg/d), auranofin (oral gold, 0.15 to 0.20 mg/kg/d), and two low dose levels of methotrexate [5MTX, 5 mg/M2/wk; 10MTX, 10 mg/M2/wk]. A total of 520 children with JRA were enrolled into these trials. Only 10MTX resulted in significantly greater improvement than placebo in variables that assess effectiveness: physician's global assessment, a composite index, and erythrocyte sedimentation rate. Treatment effect sizes were the largest in the 10MTX group for all articular disease indices. The short-term safety profiles were similar across all treatment groups. It is concluded that the current trend among pediatric rheumatologists to use oral methotrexate as the first advanced drug therapy in JRA is appropriate and that the minimum effective dose is 10 mg/M2/wk.
8481650 Iris protein antibodies in serum of patients with juvenile rheumatoid arthritis and uveiti 1993 Homogenates of bovine iris were fractionated by gel filtration chromatography, and the column-eluted proteins were probed with pooled sera obtained from patients with pauciarticular juvenile rheumatoid arthritis (JRA). The serum pool prepared from patients with pauciarticular JRA and a history of anterior uveitis, but not from those without the eye disease, contained IgG antibodies which bound a low molecular weight iris antigen (LMW-IA) as measured by a modified ELISA. LMW-IA was protease-sensitive and contained at least four proteins of approximate molecular weights of 16, 13, 9 and 6.5 kD with no uronic acid or carbohydrate. Analysis of individual patient sera for IgG anti-LMW-IA antibody demonstrated that 1/20 (5%) pediatric non-rheumatic disease controls (NRDC), 1/19 (5.3%) non-uveitic pauciarticular JRA patients and 6/21 (28.6%) uveitic pauciarticular JRA patients were positive by ELISA. Levels of anti-LMW-IA antibody did not correlate with serum IgG concentration, the presence of IgG antibody to soluble retinal S antigen (S antigen) or reactivity to the low molecular weight fraction of bovine choroid (LMW-C). Ten of 21 (47.6%) children with pauciarticular JRA and uveitis had serum antibody that reacted with LMW-IA and/or retinal S antigen as compared to 1/20 (5%) NRDC patients and 3/19 (15.8%) patients with pauciarticular JRA uncomplicated by uveitis. Analysis of patient immunoreactivity to proteins of the anterior uveal tract may provide a greater understanding of pathogenic features related to arthritis-associated eye disease.
8342606 Rabbit models of arthritis: immunolocalization of matrix metalloproteinases and tissue inh 1993 Aug The distribution of the matrix metalloproteinases, collagenase, stromelysin, gelatinases A and B, and the tissue inhibitor of metalloproteinases in cartilage and synovium removed from rabbits up to 27 days after induction of two models of arthritis was investigated by immunolocalization. Following intra-articular injection of poly-D-lysine/hyaluronic acid coacervate, collagenase and stromelysin were found bound to cartilage matrix, but there was little increase in chondrocyte synthesis of these enzymes. The synovium underwent a complex wound healing response involving invagination and encapsulation of the coacervate and inflammatory cell debris, during which all four metalloproteinases and tissue inhibitor of metalloproteinase could be immunolocalized. The second model, intra-articular injection of ovalbumin into sensitized rabbits, caused considerable chondrocyte necrosis; collagenase was found bound to cartilage matrix on day 13, although again there was little evidence of synthesis by chondrocytes. Inflammatory cell infiltration of meniscoid synovia took place initially, followed by fibrosis involving macrophagelike cells secreting gelatinase A. In both models there was rapid loss of glycosaminoglycan metachromasia from the cartilage matrix. These results are discussed in relation to current knowledge of metalloproteinase involvement in the chronic rheumatoid synovial pannus erosion of cartilage in humans. The data suggest that there are considerable differences between rheumatoid arthritis and these models, and their use must therefore be carefully defined.
8539715 [Adult-onset Still's disease. An underdiagnosed condition?]. 1995 Nov 30 Adult onset Still's disease is a variant of systemic juvenile chronic arthritis in adulthood. The clinical picture is characterized by high spiking fever, arthralgia/arthritis, transient erythema, acute-phase reaction including elevated ESR, CRP and neutrophilia, resembling acute bacterial infections. Hyperferritinaemia and hepatic dysfunction are usually present, and the patients frequently have a sore throat. Extraarticular features, such as splenomegalia, serositis and pericarditis may be parts of this disease as well. Two cases are described, who were admitted to the Department of Internal Medicine of a small Norwegian hospital. Both patients were subjected to exhaustive and laborious investigations for the purpose of disclosing malignancy and/or septicaemia. Following adequate glucocorticoid therapy, both were asymptomatic after less than a week's treatment and after five months' follow-up. Two sets of diagnostic criteria are presented, having different sensitivity, although almost equal specificity. Still's disease in the adult may be an underdiagnosed clinical entity, but should definitely be considered to be a possible differential diagnosis when investigating suspected malignancy, including lymphoma and febrile conditions suspected of septicaemia.
1340942 [Anticardiolipin antibodies in rheumatic autoimmune diseases. Correlation with clinical an 1992 Nov The prevalence of anticardiolipin antibodies (ACA) was determined using an Elisa method in sera of 60 patients with autoimmune diseases. ACA were correlated with clinical manifestations of the antiphospholipid syndrome and other serological markers of autoimmunity. Sixty three percent of sera had ACA IgG (+) and 50% ACA IgM (+). Nine patients, five of them with systemic lupus erythematosus, had a history of arterial or venous thrombosis and all had positive ACA (IgG (+) in 7 and IgM (+) in 2). In patients with rheumatoid arthritis, no association was found between the presence of ACA and thrombosis. Patients with a history of spontaneous abortion had non significantly higher levels of ACA. There was a significant correlation between ACA levels and rheumatoid factor (ACA IgG r = 0.374, ACA IgM r = 0.676), no other association was found between ACA and other autoantibodies. It is concluded that ACA are found frequently in patients with autoimmune diseases and its clinical significance may be different in patients with lupus than in patients with rheumatoid arthritis or other connective tissue diseases.