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At least 325 records · Page 18Linked to original sources
PubMed2002

Propylthiouracil-induced lupus-like syndrome developing in a Graves' patient with a sibling with systemic lupus erythematosus.

A 13-year-old girl with Graves' disease, whose younger sister had systemic lupus erythematosus, developed polyarthralgia, fever, neutropenia, hypergammaglobulinemia, and microscopic hematuria after treatment with propylthiouracil (PTU) for 2 years. Myeloperoxidase-anti-neutrophil cytoplasmic antibodies were strongly positive. Anti-single- and anti-double-stranded DNA antibodies were positive, whereas LE cells and anti-Sm antibodies were negative. PTU was discontinued and all symptoms subsided gradually. Two years later, the microscopic hematuria had disappeared completely. Both patients had the identical HLA-DR alleles (HLA-DR9). These present two cases in siblings suggest that both sisters had lupus diathesis, and that the elder sister developed a PTU-induced lupus-like syndrome.

Adolescent↗
PubMed1986

A case of hereditary angioneurotic edema associated with systemic lupus erythematosus.

A pedigree of C1 inhibitor (C1 INH) deficiency associated with positive LE cell and an elevated titer of DNA antibodies and antinuclear factor (ANF) and nephropathy was presented. The proband of this family was diagnosed as having definite systemic lupus erythematosus (SLE) after a clinical course of several year since her first visit to our hospital and because of the lack of hemolytic activity of complement (CH50), in spite of the absence of idiopathic edema, C1 INH levels were 1.2 mg/dl (3.8% NHS) determined as antigen and 65 site forming unit (SFU) (5.8% NHS) determined by hemolytic assay in her blood. Her mother and brother had characteristic idiopathic edema of her face, larynx, hand and bowel and they had low levels of C1 INH of 1.8 mg/dl (5.8% NHS) and 4.8 mg/dl (15.5% NHS) respectively in their blood. On the basis of these findings, this family was diagnosed as having a pedigree of hereditary angioneurotic edema (HANE) which is supposedly an inherited autosomal positive trait. Actually, however, the proband's serological and hematological indices became positive and progressed year by year, which implies that SLE was absent for the first several years. It might be said that this interesting clinical course indicates that SLE appeared chronologically as a hereditary deficiency in one of the complement components in this case. In concurrence with the general observation that recurrent viral infections due to the deficiency of complement components are presumed to be responsible for SLE-like disease. Her levels of several kinds of anti-virus antibodies were high. Methylprednisolone helped normalize her level of C1 INH and ameliorate the clinical course.

Adult↗
PubMedDate not supplied

Association of mixed connective tissue disease, Sjögren's syndrome and autoimmune hepatitis: report of a case.

A 51-year-old female was admitted to the hospital due to liver dysfunction. She had suffered from arthralgia, Raynaud's phenomenon, dry mouth and dry eyes. Laboratory data showed positive LE cells and anti-RNP. The diagnosis of mixed connective tissue disease (MCTD) was made according to the criteria of the international symposium on MCTD and Antinuclear Antibodies (August 1986, Tokyo). Liver biopsy showed chronic active hepatitis. The results of sialography, lip biopsy and rose-bengal test were compatible with Sjögren's syndrome (SjS). This is the report of a rare case with MCTD associated with SjS and autoimmune hepatitis.

Antibodies, Antinuclear↗
PubMed1995

[Undifferentiated connective tissue syndromes (UCTS) accompanied by laryngeal involvement and autoimmune hepatitis].

Here we report a patient with undifferentiated connective tissue syndromes (UCTS) who developed hoarseness during exacerbation of autoimmune hepatitis. A 51-year-old woman was hospitalized in November 1993 because of hoarseness and liver dysfunction. She had demonstrated Raynaud's phenomenon, polyarthralgia and hoarseness since 1992. In August 1993, liver dysfunction was noted. On admission, laboratory data showed mild leukopenia, thrombocytopenia (WBC 3,900/mm3, platelet 12.4 x 10(4)/mm3), and elevations of transaminase (GOT 96 IU/l, GPT 79 IU/l) and IgG (4,556 mg/dl). Anti-nuclear antibody (ANA) and anti-smooth muscle antibody were positive. Other autoantibodies including anti-DNA antibody, anti-Scl 70 antibody were all negative. LE test and LE cells were also negative. On laryngoscopic examination, lesions that appeared similar to a bamboo-joint were noted at the middle of the bilateral vocal cords. Pathological findings of liver biopsy specimen were compatible with autoimmune hepatitis. She was treated with 30 mg of prednisolone. Polyarthralgia, hoarseness and the abnormalities of the transaminase levels improved rapidly. Laryngoscopic findings were also normalized. We considered this laryngeal involvement to be acute laryngitis accompanied by some UCTS, including a typical systemic lupus erythematosus (SLE) because of arthritis, cytopenia and ANA positivity. Involvement of the larynx in collagen disease is rarely mentioned in published reports.

Anti-Inflammatory Agents↗
PubMed1995

[A case of SLE associated with antiphospholipid syndrome and mitral stenosis].

This case involves a 41-year-old woman with SLE. The patient began having symptoms of arthralgia in 1978 and developed fever, pleuritis and lupus psychosis in 1986. Laboratory exams showed positive antinuclear-antibody, LE-cell phenomenon, hypocomplementemia and lupus anticoagulant. Echo cardiography demonstrated mitral regurgitation and stenosis. She was treated with 50 mg of prednisolone and these manifestations subsided. In 1989, she developed dyspnea on exertion and echo cardiography revealed severe mitral stenosis. Pulmonary infarction was detected by MAA lung scintigraphy. At this time, she was diagnosed as SLE associated with antiphospholipid syndrome (APS). A mitral valvular replacement operation was performed in 1991. Pathological studies of mitral valve demonstrated Libman Sacks endocarditis. APS is known occasionally to complicate with left-sided valvular diseases, mitral stenosis is quite rare in both SLE and APS. This patient reveals a rare case of SLE associated with APS and mitral stenosis. It is suggested that this patient developed mitral stenosis with Libman Sacks endocarditis, associated with the presence of antibody against phospholipids.

Adult↗
PubMed1999

[Crohn's disease with the onset resembling systemic lupus erythematosus].

We described a 37-year-old man with Crohn's disease (CD) resembling systemic lupus erythematosus (SLE) at his disease onset. He was admitted to the municiple Akiru Hospital in October 1986 by fever, aphtous oral ulcerations, sore throat and polyarthralgia. Hematologic examination showed leukocytopenia, lymphocytopenia, positive tests for antinuclear antibody, anti-DNA antibody and LE cell phenomenon. He has had episodes of convulsion and conciousness loss of unknown etiology when he was 17 years old. The diagnosis of SLE was made, and oral medication of prednisolone was started. Several weeks later, most of symptoms and autoantibodies disappeared, although the oral aphtous ulcerations and leukocytopenia remained. In May 1987, he admitted to the other hospital because of bloody vomiting. Endoscopic examination showed the esophagial ulceration, and histology of biopsied-specimen was nonspecific esophagitis. The combination of prednisolone and oral cyclophosphamide or methotrexate was employed thereafter. However, the leukocytopenia, oral aphtous ulceration and esophagial ulceration continued in spite of these treatments. All the immunosuppressive treatment was stopped at March 1992. In October 1995, he admitted to our hospital because of body weight loss and continuous diarrhea with occasional bloody stool. Barium enema and endoscopic examination of the colon revealed the findings compatible with CD. The patient responded favorably to methylprednisolone pulse therapy followed by oral sulphasalazine. This case indicated that cases with inflammatory bowel diseases like CD could show similar clinical signs and symptoms to SLE, and in some cases of CD might satisfied the classification of criteria for SLE.

Administration, Oral↗
PubMed1984

Systemic vascular lesions and prognosis in systemic lupus erythematosus.

The purpose of this paper is to show the relationship of systemic vascular lesions to the clinical manifestations and prognosis in SLE. Thirty-four autopsied cases of SLE formed the subject material for this study. Histopathological tissue studies were made on vascular lesions from almost all organs. Vascular lesions were separated into three groups according to the size of the involved vessel: 1) medium-sized artery (9 cases), 2) small artery (12 cases) and 3) (13 cases) without systemic vascular lesions. Vascular lesions were also separated into five groups (a) fibrinoid degeneration (10 cases), b) intimal thickening (6 cases), c) thrombosis (6 cases), d) sclerosis (7 cases) and e) (13 cases) without systemic vascular lesions. Patients with involvement of medium-sized arteries had a low female incidence, photosensitivity, and positive LE cell incidence, and the cause of death in these patients was cerebral vascular involvement. The cause of death in patients with vascular fibrinoid degeneration and thrombosis was mainly uremia, whereas patients with sclerosis more often died from infection. Regarding systemic vascular lesions in SLE, the prognosis for the patients with thrombosis and vascular involvement of medium-sized arteries was the most grave.

Adult↗
PubMed1990

Two cases of suspected arteriosclerotic optical chiasmal syndrome.

Optic chiasmal syndrome due to arteriosclerotic vascular changes is rate. To our knowledge, there has been no report of arteriosclerotic chiasmal syndrome in Korea. In our two cases, other causes of chiasmal syndrome were not detected by MRI, four-vessel cerebral angiography, CSF study, and special laboratory examinations including ANA test, RA factor, and LE cell. With conservative treatment, the visual fields of the two patients are showing progressive improvement. We report here two cases of suspected arteriosclerotic optic chiasmal syndrome.

Adult↗
PubMed1977

Less common side effects of methyldopa.

A patient with methyldopa-induced hyperpyrexia who also developed positive antinuclear factors, LE cells and ocular disturbance is described. These side effects disappeared when the drug was withdrawn. The incidence of these side effects is reviewed. Methyldopa should be withdrawn when these side effects appear, as less troublesome antihypertensive drugs are now readily available.

Aged↗
PubMed1999

Young age at onset, renal involvement, and arterial hypertension are of adverse prognostic significance in juvenile systemic lupus erythematosus.

OBJECTIVE: To look for associations between mortality, clinical or laboratory data, and age at disease onset in systemic lupus erythematosus patients aged 16 years or younger at disease onset. PATIENTS AND METHODS: The medical records of patients seen at the Clinics Hospital, State University of Campinas, São Paulo, Brazil, between 1979 and 1995 were reviewed retrospectively. All 59 included patients (48F/11M) fulfilled four or more American College of Rheumatology criteria for systemic lupus erythematosus. Patients with discold, drug-induced or neonatal lupus, or other systemic connective tissue diseases were excluded. Patients were studied individually then classified in three groups based on age at disease onset, as follows: Group I, < or = 9 years of age; Group II, 10-14 years of age; and Group III, 15-16 years of age. Clinical and laboratory abnormalities and mortality were compared in the three groups. RESULTS: The most frequent clinical manifestations were joint symptoms (91.5%), renal involvement (71.1%), malar rash (61%), alopecia (61%), fever (59.3%) and photosensitivity (52.5%). Laboratory findings included antinuclear antibody in 94.9% of cases. LE cells in 71.1%, low serum complement in 65.3%, anti-DNA in 63.4%, hematuria in 62.7%, and proteinuria in 61%. The mortality rate was 23.7% (9F/5M) overall, 18.7% in females, and 45.4% in males (P = 0.07). The cause of death was infection in eight patients (57.1% of decedents), central nervous system disease in five (35.7%), and renal insufficiency in one (7.2%). Disease onset before 15 years of age (P = 0.026), renal involvement (P = 0.03), and arterial hypertension (P = 0.002) were predictive of mortality. Mortality was not influenced by gender or race.

Adolescent↗
PubMedDate not supplied

Endoscopic pericardial fenestration for a patient with sustained lupus pericarditis.

A 57-year-old woman was diagnosed in January 1982 with SLE based on ANA 1:640, positive LE cell preparation, proteinuria (3+), and pericarditis. In 1984, 1994, and 1997, the pericardial effusion was noted to have increased without signs of disease exacerbation or cardiac tamponade, and pericardial drainage was repeated to control the effusion. A massive pericardial effusion developed in August 1997. After tuberculosis, hypothyroidism, neoplasm, and progression of SLE were ruled out, we decided to perform pericardial fenestration. A safe and minimally invasive pericardial fenestration was successfully completed endoscopically. Pathologic study of the specimen revealed chronic pericarditis. We consider endoscopic pericardial fenestration to be useful for at risk patients with pericarditis to control the effusion and establish a differential diagnosis.

Endoscopy↗
PubMed1999

Clinical profile of polymyositis in Kashmir valley.

OBJECTIVES: The present study is a retrospective and prospective study of 24 cases of idiopathic inflammatory myopathies from Kashmir valley. The study was conducted to depict the clinical profile of polymyositis from this part of the world. METHODS: The diagnosis of polymyositis/dermatomyositis was established by following the criteria of Bohan and Peter (1975). Besides relevant clinical examination, investigations like complete blood count, ESR, muscle enzymes, LE cell phenomenon, antinuclear antibodies and rheumatoid factor were also done. Electrophysiological study and open muscle biopsy was performed in 21 and 24 cases respectively. RESULTS: The mean age at presentation was 34 years with 62.5% cases presenting in fourth decade. Pelvic girdle weakness was observed in all the 24 cases. Male:Female ratio was 1:1.4. Four (16.8%) cases had associated collagen vascular disease. No case was associated with malignancy or childhood vasculitis. Raynauds' phenomenon was seen in seven (29.1%) cases. Creatine phosphokinase and lactic dehydrogenase was elevated in 22 (91.6%) and 12 (50%) cases respectively. Electromyography revealed myopathic features in 74.3% cases. Muscle biopsy revealed features of inflammatory myopathy in 22 (91%) cases. CONCLUSIONS: The conclusions drawn from this study are as follows: a. Younger age at presentation b. Shorter duration of illness at presentation c. Increased frequency of Raynaud's phenomenon as compared to other Indian series. d. No case of polymyositis/dermatomyositis associated with malignancy or childhood vasculitis was seen. e. Biochemical, electrophysiological and histopathological features suggestive of shorter duration of illness were observed.

Adolescent↗
PubMed2001

[A case of lupoid hepatitis satisfying criteria for systemic lupus erythematosus associated with psychosis].

A 26-year-old female was admitted to our hospital because of arthralgia and general fatigue. On examination, she had malar rash and arthritis. Laboratory data revealed AST, ALT and gamma-globulin elevation, antinuclear antibody and double-stranded DNA antibody positivity, and LE cell phenomenon. Liver biopsy showed marked lymphocytic infiltration and slight fibrosis in the portal areas. She was diagnosed with lupoid hepatitis, and also satisfied the criteria for SLE including malar rash, arthritis, immunologic disorder and antinuclear antibody. She was administered prednisolone, after which AST and ALT decreased. She developed psychosis and her electroencephalogram showed diffuse slow waves corresponding to psychosis by SLE. Lupoid hepatitis is frequently associated with various systemic manifestations. However, only a few cases of lupoid hepatitis satisfying the criteria for SLE associated with psychosis have been reported.

Adult↗
PubMed1992

[Myxoma of the left atrium suggesting a connective tissue disease].

A case is presented of myxoma of the left atrium suggesting a connective tissue disease. In the foreground were musculo-articular symptoms, sub-febrile states, considerable loss a body weight. At the same time an increased sedimentation rate, hypergammaglobulinaemia, increased level of IgG and IgM, high CRP level, and the presence of antinuclear antibodies and LE cells were noted. The diagnosis of myxoma was made on the basis of echocardiographic examination. After surgical resection of the tumor all symptoms regressed, and a normalization was observed of the formerly observed abnormalities in the laboratory investigations.

Adult↗
PubMed1992

[A case of Fabry's disease associated with lupus nephritis].

A 36-year-old woman was hospitalized because of nephrotic syndrome. On admission, laboratory studies revealed total protein 5.9g/dl, total cholesterol 381mg/dl, urine protein 2-4g/day, C3 68mg/dl(90-185mg/dl) and the immunological tests showed that antinuclear factor, anti-DNA antibodies and the LE cell phenomenon were positive. Renal function was within normal range. After admission, renal biopsy was done. Light microscopic finding showed diffuse membranous glomerulonephritis, and vacuolization of epithelial cells. Immunofluorescent microscopic finding showed a granular specific staining for IgG, IgM, C3 and C1q along the capillary loops. Electron microscopic finding showed subepithelial and subendothelial dense deposits, and visceral epithelial cell cytoplasm containing osmiophilic multilamellar lipoid bodies. In the studies of the enzyme activities, the patient's fibroblast extract demonstrated a partial deficiency of alpha-galactosidase, and urine ceramide trihexoside was positive. But the patient's leukocyte extract did not demonstrate a deficiency of alpha-galactosidase. So Fabry's disease associated with lupus nephritis was diagnosed. It seems that the case of Fabry's disease which is an X-linked disorder caused by deficiency of the lysosomal enzyme alpha-galactosidase, associated with lupus nephritis, is extremely rare.

Adult↗
PubMed2004

Myocarditis as an initial manifestation of systemic lupus erythematosus.

We report a case of systemic lupus erythematosus (SLE) whose initial presentation was in the form of myocarditis. The patient did not have arthritis, fever or butterfly rash. Presence of LE cell phenomenon, positive ANA, anti-DS DNA antibodies, leucopenia and high ESR with polyserositis indicated the diagnosis to be SLE. Therapy with steroid resulted in complete recovery. The patient developed atrial fibrillation during her course of acute illness which is being reported for the first time.

Adult↗
PubMed2004

Lupus band test in systemic lupus erythematosus.

The usefulness of the direct immunofluorescent antibody technique--lupus band test--for the diagnosis of systemic lupus erythematosus (SLE) has been well established. The aims of the study were to determine the prevalence of the LBT at various sites of the skin in a cross section of patients with SLE and its correlation with disease activity. The LBT was demonstrated in 64% of skin lesions, 63% in non-lesional sun-exposed (NLSE) skin and 25% in non-lesional sun-protected (NLSP) skin. The prevalence of the LBT in lesional and NLSE groups was significantly different from the NLSP group (p = 0.03 and 0.005 respectively). There was a significant correlation between the presence of a positive LBT in NLSE skin with the presence of the LE cell phenomenon (p = 0.04) and anti - ds DNA antibody (0.02). In addition, there was a significant correlation between IgG LBT in the NLSE skin with serum hypocomplementaemia (p = 0.03) and anti - ds DNA antibody (p = 0.04). Other than these, no significant correlation was detected between the LBT from the 3 sites with overall clinical activity, renal disease, active skin lesions, or other laboratory indices of activity. These findings suggest that the LBT is mainly indicated as a diagnostic tool and has little role in assessing disease activity.

Adolescent↗
PubMed1992

[A case of myocardial infarction in a young man with systemic lupus erythematosus].

Apparently the incidence of coronary artery disease in systemic lupus erythematosus (SLE) has been increasing. However, most of the cases had been treated with corticosteroids, and had atherosclerotic lesions in the coronary arterial tree. A 21-year-old man with latent and untreated SLE had an attack of acute myocardial infarction. Coronary arteriography showed eccentric stenotic lesion at the proximal segment of the right coronary artery. One week later, in the 2nd coronary arteriography, this stenotic lesion was not able to be recognized. We supposed that the coronary artery occlusion was due to thrombus formation, and was not related to atherosclerosis, arteritis and embolus. He had no coronary risk factors. Laboratory data showed lymphocytopenia, proteinuria, positive antinuclear antibody, and positive LE cell, and the case was diagnosed as SLE. Subsequent investigations showed the presence of antibodies to cardiolipin. It was suggested that anticardiolipin antibody and other thrombogenic factors were the causes of the coronary occlusive thrombosis in this patient with SLE.

Adult↗