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ID PMID Title PublicationDate abstract
11299055 Angiogenesis in the pathogenesis of inflammatory joint and lung diseases. 2001 This paper reviews hypotheses about roles of angiogenesis in the pathogenesis of inflammatory disease in two organs, the synovial joint and the lung. Neovascularisation is a fundamental process for growth and tissue repair after injury. Nevertheless, it may contribute to a variety of chronic inflammatory diseases, including rheumatoid arthritis, osteoarthritis, asthma, and pulmonary fibrosis. Inflammation can promote angiogenesis, and new vessels may enhance tissue inflammation. Angiogenesis in inflammatory disease may also contribute to tissue growth, disordered tissue perfusion, abnormal ossification, and enhanced responses to normal or pathological stimuli. Angiogenesis inhibitors may reduce inflammation and may also help to restore appropriate tissue structure and function.
11242226 A comparative study between MR sialography and salivary gland scintigraphy in the diagnosi 2001 Mar PURPOSE: The purpose of this work was to compare the diagnostic accuracy of MR sialography with that of salivary gland scintigraphy in Sjögren syndrome. METHOD: One hundred thirty patients clinically suspected of having Sjögren syndrome were examined by MR sialography and salivary gland scintigraphy. A labial gland biopsy was performed in all patients. Imaging findings of MR sialography and salivary gland scintigraphy were compared with the results of labial gland biopsy. RESULTS: From the results of labial gland biopsy, the diagnosis of Sjögren syndrome was established in 80 patients. Abnormally high T2 signal intensity areas on MR sialography and decreased uptake and delayed excretion of [(99m)Tc]pertechnetate on salivary gland scintigraphy were well seen in patients with Sjögren syndrome. For the diagnosis of Sjögren syndrome, salivary gland scintigraphy showed higher sensitivity than MR sialography. On the other hand, MR sialography showed higher specificity and positive predictive value (PPV) than salivary gland scintigraphy. Overall diagnostic accuracy was 83% for MR sialography and 72% for salivary gland scintigraphy. CONCLUSION: The high PPV of MR sialography suggests that MR sialography is the preferred imaging modality in patients suspected of having Sjögren syndrome.
10086903 Multisystem neuronal involvement and sicca complex: broadening the spectrum of complicatio 1999 Mar We report two patients with multisystem neuronal involvement associated with sicca complex. One had a lower motor neuron syndrome combined with a flaccid bladder and rectum. The second patient had unilateral hearing loss, sensory neuronopathy, Adie's pupils, upper motor neuron signs, and autopsy-proven anterior horn cell degeneration. Our cases lead us to propose that the spectrum of neuronal involvement occurring with sicca syndrome may be wider than is currently appreciated.
9935059 Quantitative evaluation of salivary gland scintigraphy in Sjörgen's syndrome. 1999 Jan This study compared the quantitative characteristics of salivary gland scintigraphy in patients with Sjogren's syndrome with the histopathologic grading of labial biopsy. METHODS: Thirty-nine patients with Sjögren's syndrome were studied by salivary gland scintigraphy and labial biopsy. Twelve normal volunteers were also studied as a control group for scintigraphic parameters. After injection of 370 MBq 99mTc sodium pertechnetate, dynamic salivary scintigraphy with lemon juice stimulation was performed for 50 min. Functional parameters for the parotid and submandibular glands were calculated, and scintigraphic and histopathologic results were compared. RESULTS: With the progression of histopathologic grades 1-4, the velocity of tracer secretion decreased in the parotid gland (P < 0.05), and the quantity of tracer accumulation decreased in the submandibular gland (P < 0.05). The histopathologic grade in patients with Sjögren's syndrome was correlated with these scintigraphic parameters (P < 0.05): Histopathologic grade = 2.304 - 0.014 x (submandibular gland maximum accumulation) + 0.196 x (parotid gland time interval of tracer secretion). CONCLUSION: Decreased secretion velocity in the parotid gland and decreased accumulation in the submandibular gland were sensitive indicators of salivary gland disease in Sjögren's syndrome. The histopathologic grade was correlated with these scintigraphic parameters.
9775130 [Adult-onset Still's disease after hepatitis A and B vaccination?]. 1998 Feb BACKGROUND: Hepatitis A and B vaccination are generally very well tolerated. However, exceptional cases of arthritis and systemic diseases have been reported after hepatitis B vaccination. CASE REPORT: The authors report a case of adult Still's disease apparently triggered by hepatitis A and B vaccination. The patient was a 38-year-old woman who presented with fever, hepatitis, pneumonitis and neurologic symptoms, compatible with the diagnosis of adult Still's disease. DISCUSSION: The authors discuss the rarity of systemic diseases triggered by such vaccination, the link between hepatitis B virus and some vasculitis, and the possibility for viral bacterial infections to trigger adult Still's disease.
9350172 A superfemale with primary Sjögren's syndrome which involved systemic organs. 1997 Sep A 52-year-old Japanese woman complicated by a sex chromosomal anomaly as a superfemale, a mosaic of XXXXX/XXXX/XXX/XX/XO, with mild mental retardation, was hospitalized for dry mouth, dry eyes, and proteinuria. The sialography of the right parotid gland showed a globular-type gland enlargement. A definite diagnosis of primary Sjögren's syndrome (SS) was made, and further examinations revealed not only typical sicca syndrome but also systemic extraglandular lymphocytic infiltration; interstitial pneumonitis, glomerular- and interstitial nephritis, superficial gastritis, thyroiditis, and a severe excitation conductive impairment of heart. We report a very rare case of superfemale with primary SS which involved systemic organs.
9109867 Peripheral neuropathy associated with sicca complex. 1997 Apr Peripheral neuropathy occurs in Sjögren's syndrome, a disorder in which systemic immunologic phenomena, including vasculitis, are common. Neuropathy also occurs with isolated sicca complex (keratoconjunctivits sicca and xerostomia); whether this represents a distinct syndrome is unclear. We retrospectively studied 54 patients with sicca complex and peripheral neuropathy to determine mode of presentation, neuropathic patterns, frequency and pattern of serologic abnormalities, and frequency of systemic disease, including necrotizing vasculitis. Peripheral neuropathy was the presenting problem in 87%. Although sicca symptoms occurred in 93%, they were a presenting complaint in only 11% and were usually mild, reported only after specific inquiry. Minor salivary gland biopsy was positive in 73%. Sensory neuropathies strongly predominated; 61% of patients manifested either sensory polyneuropathy or polyganglionopathy. Less common patterns included sensorimotor polyneuropathy (17%) and polyradiculoneuropathy (11%). Vasculitic neuropathy was demonstrated in only two patients, but nonspecific epineurial inflammation was present in 70% of nerve biopsies. Clinical evidence of systemic disease was uncommon, particularly in the sensory polyganglionopathy group, in whom extraglandular features other than weight loss occurred in only 1 of 12 patients. Antibodies to extractable nuclear antigens, the most specific serologic marker of Sjögren's syndrome, were present in 10.4%. We conclude that peripheral neuropathy and isolated sicca complex form a distinctive syndrome in which neuropathy is the presenting feature and sicca is easily overlooked; sensory polyneuropathy and polyganglionopathy predominate; serology is confirmatory but very insensitive; and extraglandular disease, including vasculitis, is uncommon compared with typical Sjögren's syndrome. Tests of ocular or salivary involvement are needed for diagnosis, and demonstration of inflammation in biopsied nerve is supportive. Improved definition of this disorder should permit further studies of natural history and efficacy of immunotherapy.
9826056 Ischemic choroidopathy in primary Sjögren's syndrome. 1998 Oct PURPOSE: The authors describe a 68-year-old woman in whom ischemic choroidopathy and optic neuropathy developed in association with primary Sjögren's syndrome with central nervous system involvement. METHODS: Diagnosis of primary Sjögren's syndrome was made upon the association of keratoconjunctivitis sicca, minor salivary gland biopsy and serologic abnormalities. Fluorescein angiography showed signs highly suggestive of ischemic choroidopathy. The authors discuss differential diagnosis and pathophysiology of such choroidal manifestations. Treatment consisted in massive steroid therapy. Secondarily, an immunosuppressant agent (azathioprine) was successfully added because of neurological recurrences. CONCLUSION: Primary Sjögren's syndrome should be considered in the differential diagnosis of ischemic choroidopathy.
11412873 Sensory neuropathy of the trigeminal, glossopharyngeal, and vagal nerves in Sjögren's syn 2001 May 1 Isolated cranial nerve involvement in primary Sjögren's syndrome (primary SS) has rarely been described. We report the case of a patient with sensory neuropathy of the trigeminal and also the glossopharyngeal and vagal nerves, which has not been identified previously. The electrophysiological findings in our patient with primary SS confirmed trigeminal sensory neuropathy with abnormal blink reflexes and abnormal cutaneous masseter inhibitory reflexes.
10771569 [A male case of Sjögren's syndrome presenting with generalized lymphadenopathy and swelli 2000 Feb A 68-year-old man noticed severe oral dryness and a submandibular swelling. Sjögren's syndrome (SS) was diagnosed based on microscopic findings of a labial salivary gland biopsy although both anti-SS-A and anti-SS-B antibodies were negative. In addition, hypergammaglobulinemia (IgG 7940 mg/dl) and hypocomplementemia were pointed out and he was admitted to our department. On admission cervical, mediastinal, and abdominal lymph nodes swelling were detected together with enlargement of lacrimal and salivary glands. Lymphoproliferative disorders associated with SS were highly suspected. Biopsied specimens of his lacrimal gland and cervical lymph node disclosed neither malignant cells nor monoclonal proliferation of lymphocytes. An administration of corticosteroids caused rapid diminution in size of lacrimal glands, salivary glands, and lymph nodes. Both lacrimation and salivation recovered, and hypergammaglobulinemia and hypocomplementemia returned to normal after treatment. The characteristics of this case were an atypical onset in an elderly man, the negativity of anti-SS-A and anti-SS-B antibodies, and reversibility of dryness by corticosteroid treatment. These findings suggest that the pathogenesis of this case may be different from typical SS.
9489815 Establishing guidelines for the role of minor salivary gland biopsy in clinical practice f 1998 Feb OBJECTIVE: To evaluate the predictive value of clinical variables for the finding of a positive minor salivary gland biopsy (focus score > or = 2) in patients investigated for Sjögren's syndrome (SS). METHODS: One hundred twenty-one patients with sicca symptoms were referred to a multidisciplinary SS clinic in a tertiary hospital. Each patient was evaluated on protocol and labial salivary gland (LSG) biopsy was obtained. Using the San Diego criteria as a model, patient data were subjected to a cross sectional analysis on an algorithm to determine when the LSG biopsy would be most useful for determining the diagnosis of SS in clinical practice. RESULTS: Eighty-four patients had sufficient data to be included in the study. Forty patients had LSG biopsy with focus score < 2 and 44 had focus score > or = 2. Twenty-three patients had objective evidence of sicca and positive serology according to criteria standards. Eighteen of these had a positive biopsy (78%). The remaining 5 patients had many extraglandular features suggestive of SS, and the biopsies appeared to add little practical information. Patients with incomplete criteria for sicca could be diagnosed as possible SS (3 of 4 criteria) with a positive biopsy in 14 of 18 cases. The finding of anti-Ro or anti-La positivity in patients with incomplete criteria for sicca predicted a positive LSG biopsy in 85.7% of such cases. Patients with incomplete sicca and negative anti-Ro and anti-La had a negative LSG biopsy in 82% of cases. CONCLUSION: The LSG biopsy is most necessary in patients who have partial San Diego criteria for sicca and positive anti-Ro or anti-La antibody. Where SS is not reasonably suspected, or where the diagnosis is clinically obvious, the LSG biopsy adds little useful clinical information.
9287375 Abnormalities in skeletal growth in children with juvenile rheumatoid arthritis. 1997 Aug A review of the acquisition of peak skeletal mass in normal children and studies that have been reported for children with JRA lead to the following tentative conclusions: (1) The appendicular skeleton is predominantly the overall status of skeletal mineralization; (2) a failure to develop adequate bone mineralization is virtually universal in children with JRA and is characterized by a failure of bone formation. A failure to undergo the normal increase in bone mass during puberty is common in children with JRA and markedly decreases their potential to achieve an adequate peak skeletal mass; (3) the onset of accelerated skeletal maturation with puberty is a critical period of potential intervention in JRA. Conversely, therapeutic interventions later during adolescence offer less promise of reversal of inadequate bone mineralization; and (4) the most important therapeutic maneuver is likely to be control of the inflammation process, although there is hope, at present unsubstantiated, that supplemental dietary calcium and vitamin D, and normalization of physical activity, many lead to some "catch-up" mineralization.
11501878 Dental caries and dental health behavior of patients with primary Sjögren syndrome. 2001 Jun This comprehensive study was carried out to describe and analyze the oral health status, perceived oral health problems, patients' costs, and oral health behavior in a group of patients with primary Sjögren syndrome (PSS). In particular, the objective of this report was to assess whether Sjögren syndrome patients had more dental caries experience than a control group. Data were collected by means of interviews and clinical oral examinations. The study comprised 53 patients with PSS and a control group of 53 persons matched by age. Among the younger patients the number of decayed, missing, or filled teeth (DMFT) was 22.3, compared with 18.8 among controls (P< 0.05). In parallel, the DMFT in the old-age PSS patients was 26.2, against a DMFT of 22.1 for controls (P< 0.001). On average, the young patients had seven teeth missing, whereas two missing teeth were found among controls (P < 0.01). PSS patients had more frequent dental visits--every 3-4 months (40%)--than controls (19%). In parallel, 78% of the PSS patients brushed their teeth more than twice daily, compared with 28% of the control group. The PSS patient group reported having had more teeth extracted, more trouble with their teeth during lifetime, and higher expenses for dental treatment than controls. In spite of the more regular oral health care practices than the general population, PSS patients had experienced more dental caries and more radical dental treatment. It is suggested that the National Health Insurance should give emphasis to preventive care to patients with PSS.
10432943 [Adult-onset Still's disease. Differential diagnosis in recurrent pharyngitis, fever of un 1999 Jul 9 HISTORY AND PHYSICAL EXAMINATION: Six weeks before admission a 43-year old previously healthy woman got right-sided pharyngitis, accompanied with Herpes labialis and oral candidiasis. Two weeks later she travelled to a holiday in the Caribbean. During the first week of holiday she developed pharyngitis again, this time accompanied with fever and arthralgies. Therapeutic trial with antibiotics, antimalaric drugs and antiamoebics, given at the holiday resort, did not reduce fever. Immediately after return to Germany a therapeutic trial with imipenem was was attempted, without any apparent improvement. At admission, the patient was febrile, had pinky patchy exanthema and arthralgias. EXAMINATIONS: At admission, abnormal findings included anaemia (Hb 8.8 mg/dl), severe leukocytosis (35.3/nl), increased ESR (43/89 mm), CRP (24.2 mg/dl) and ferritin (5751 micrograms/l). Ultrasound examination revealed mild splenomegaly. Computed tomography of the abdomen and chest were without apparent abnormalities. TREATMENT AND CLINICAL COURSE: Infection, autoimmune diseases and neoplasia were ruled out. The diagnosis of adult onset Still's disease was established on the basis of a typical triad of symptoms (fever, exanthema, arthritis). Treatment with 100 mg/d prednisolon (started intravenously) was beneficial for fever and arthralgia, and resulted in the normalisation of laboratory findings. After gradual reduction in the corticosteroid dosage, maintenance treatment with 20 mg/d prednisolon was continued over following months. CONCLUSION: Recurrent prodromal pharyngitis and excessive hyperferritinaemia are, in addition to the triad fever-exanthema-arthritis, further important diagnostic criteria of adult onset Still's disease.
9563275 Spongiotic annular erythema in SS-A/SS-B antibody negative Sjögren's syndrome. 1998 Feb We reported four cases of Sjögren's syndrome (SjS) who manifested a new type of annular erythema that differs from the previously described annular erythema seen in anti-SS-A/SS-B antibody positive SjS in both clinical and histological findings. Characteristic histological features are the presence of spongiotic changes around the acrosyringium and perivascular lymphocytic infiltration without liquefaction degeneration or epidermal change, suggesting lupus erythematosus. No complement or immunoglobulin depositions are demonstrated along the basement membrane zone or around blood vessels. Clinically, this type of erythema usually appears on the trunk or extremities with itchy sensations, especially in summer, which contrasts with the preferential occurrence of the previously reported SjS related annular erythema on the facial skin in winter. Immunologically, all four cases lacked anti-SS-A and anti-SS-B antibodies, but possessed positive anti-microsome antibodies or thyroid tests. In three cases, metal allergy was demonstrated by patch test, which might suggest that the sweat duct is the primary target of excreted metals in this condition and that underlying SjS might play some role in the higher prevalence of metal allergy or in induction of sweat duct injury, similar to the interstitial nephritis which is now thought to be an exocrine manifestation of SjS.
9550600 Primary Sjögren's syndrome complicated by sarcoidosis. 1998 Feb Sjögren's syndrome and sarcoidosis share several common features, such as keratoconjunctivitis sicca, swelling of parotid glands, lung involvement, cutaneous anergy, T cell-mediated immunodeficiency, an increased CD4+/CD8+ lymphocyte ratio, and association with the human leucocyte antigen (HLA)-B 8 and DR 3 haplotypes. However, only five patients with primary Sjögren's syndrome and sarcoidosis have been previously reported in the English language literature. The rare case of a 49-year-old Japanese woman with primary Sjögren's syndrome complicated by sarcoidosis is described. The serum angiotensin-converting enzyme level was increased, and histological examination of lung and skin biopsies revealed noncaseating granulomas, indicating that her primary Sjögren's syndrome was complicated by sarcoidosis.
9175937 Subgroups of primary Sjögren's syndrome. Sjögren's syndrome in male and paediatric Greek 1997 May OBJECTIVES: To describe the clinical and serological findings in male and paediatric Sjögren's syndrome (SS) patients. PATIENTS AND METHODS: Using the European criteria for the diagnosis of SS 12 male and 13 paediatric patients were identified and compared with those of 30 consecutive unselected adult female SS patients. RESULTS: The mean (SD) age of paediatric patients was 9.4 (2.2) years, ranging from 6 to 14 years. Recurrent parotid gland enlargement was the initial clinical manifestation in the majority of the children with a statistical significance compared with male (p < 0.01) and with female patients (p < 0.0001). Sicca manifestations were the most common clinical symptoms in male and female patients at disease onset. The systemic manifestations were similar among the three groups except that men showed lower frequency of arthritis (p < 0.05) and Raynaud's phenomenon (p < 0.05) compared with women. No differences were found among the immunological profile of children and female patients, while male patients had a lower frequency of antinuclear antibodies (p < 0.025) and antibodies to Ro(SSA) nuclear antigens (p < 0.025) compared with women. CONCLUSION: Primary SS is rare in children and men in Greece. Recurrent parotid gland enlargement is the most common clinical finding at disease onset in children. Male patients seem to have less systemic manifestations and lower frequency of autoantibodies.
9146077 [A case of Sjögren's syndrome with a high titer of anticardiolipin antibody that develope 1997 Jan A 50-year-old-male consulted us, complaining of gait disturbance. He was diagnosed as having Parkinson syndrome, but had no benefit of drugs for Parkinson disease. On admission he showed symptoms of parkinsonism such as parkinsonian gait, bradykinesia and muscle rigidity, and recurrent aseptic meningitis. Additionally, his condition was complicated by Sjögren's syndrome, based on the preliminary criteria of Sjögren's syndrome established by the European Community. Because his parkinsonism and meningitis were considered to be caused by Sjögren's syndrome, he was administered corticosteroids, which improved his parkinsonism and aseptic meningitis. The brain MR images in our case did not match those typically reported in cases of central nervous system Sjögren's syndrome (CNS-SS). Although the most common abnormalities are small distinct areas of increased signal intensity on T2-weighted images, the MR images in our case revealed periventricular diffuse areas of increased signal intensity on T2-weighted images. Although his parkinsonism and aseptic meningitis are believed to the effects of vasculitis caused by Sjögren's syndrome, it is also possible that his parkinsonism is related to Binswanger's disease resulting from a high titer of anticardiolipin antibody. Our report proposes a possible relationship between anticardiolipin antibody and CNS-SS when MR images reveal diffuse high intensity areas on T2-weighted images.
10680194 Apheresis. 2000 Feb The removal of pathologic humors by various methods is an ancient medical remedy used in the management of diseases whose pathophysiology is poorly understood and whose effective treatment modalities are lacking. The contemporary means for such an approach involves apheresis, which is now possible due to advances in blood banking technologies. Apheresis has been used in most of the major rheumatic diseases, in particular systemic lupus erythematosus and rheumatoid arthritis. Although numerous case reports describe clinical benefits of apheresis in rheumatologic disorders, data from clinical trials are discouraging and suggest a limited role for apheresis in rheumatic disease management.
10090170 Reliability and sensitivity of diagnostic tests for primary Sjögren's syndrome. 1999 Mar OBJECTIVE: To investigate whether diagnostic tests for primary Sjögren's syndrome (pSS) are reproducible when repeated after one year (reliability). To evaluate whether the sensitivity of the diagnostic tests increases with repeated testing. METHODS: A structured interview investigating the subjective sensation of dry eyes and dry mouth, and the diagnostic tests Schirmer I, unstimulated whole saliva collection (UWSC), serological tests for antinuclear antibodies (ANA), for anti-Ro/SSA and anti-La/SSB antibodies as well as Waaler's test for rheumatoid factor, were performed twice with a one year interval in 66 patients with pSS. Reliability was given as the percentage of positive tests remaining positive at the second examination, while sensitivity was given as the percentage of patients with positive tests. RESULTS: Highest reliability was obtained for the sensation of dry mouth (98.2%) and sensation of dry eyes (96.4%), and anti-SSA/SSB antibodies (93.3%). Lowest reliability was obtained for rheumatoid factor at cutoff titer 1:32 (70.6%) and positive Schirmer I in one eye (77.4%). The reliability for ANA was 80% at cutoff titer 1:32, and increased to 93.3% at cutoff titer 1:128. UWSC had a reliability of 84.2%. The pooled sensitivity for all the tests increased significantly (p < 0.05) compared to the examination, which had the lowest sensitivity. CONCLUSION: The diagnostic tests for pSS are generally highly reliable when performed twice with a one year interval. The gain in sensitivity by repeating the tests is limited, being most marked for Schirmer I.