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PubMed1980

[Systemic lupus erythematosus and dermatomyositis in children].

The authors study the diagnosis and the prognosis in 25 children with systemic lupus erythematosus and in 22 children with dermatomyositis. The most frequent signs when the diagnosis is established are articular involvements, erythema of the face, renal disorders and the presence of LE cells. The symptoms are often misleading, which explains the long delay between the first sign of the disease and the time of diagnosis, which can vary between one month to nine years. The prognosis of this disease has improved because of modern treatments : only four patients died, three of them because of renal failure and one because of cerebral embolism, after six months, one year, three years and ten months, In dermatomyositis the most frequent signs at the time of diagnosis are skin involvements and muscular and articular symptoms. The period of time between the onset of the disease and the diagnosis is shorter : one month to four years, 19 times equal to or less than one year, because the clinical picture is quite clear. Ten times the clinical evolution lead to a complete cure, 9 times a clear improvement with sequelae was observed : retractile myelosclerosis, calcinosis and medullary compression. One patient died of intestinal ulceration and two of pneumonia aggravated by involvement of the respiratory muscles.

Adolescent↗
PubMed1978

Diagnosis and management of systemic lupus erythematosus.

Systemic lupus erythematosus (SLE) is not a rare disease. There are several common clinical signs which should alert the physician to a possible diagnosis of SLE and which should condition him to look for specific clinical and laboratory findings. In addition to simple screening tests, useful procedures include a search for antinuclear antibodies, lupus erythematosus (LE) cells, anti-DNA antibodies and low serum complement. Management is determined by the type of course encountered but most patients will do well under the care of their family physician.

Adult↗
PubMed1994

[Side effects of interferon on endocrine and respiratory system in 545 cases of chronic hepatitis C].

We investigated the side effects of interferon (IFN) on the endocrine and respiratory system in 545 cases of chronic hepatitis C. Eleven of 494 (2.2%) patients with chronic hepatitis C who were treated with natural or recombinant interferon (IFN) developed thyroid disease while on treatment. Eight patients developed hyperthyroidism and 3 patients developed hypothyroidism. All 11 patients required definitive therapy, who became euthyroid after the therapy. Two patients received nIFN alpha and one patient received rIFN alpha 2b developed diabetes mellitus. Two patients received rIFN alpha 2a and rIFN alpha 2b, respectively, developed interstitial pneumonia 12 weeks and 24 weeks later, respectively. One patient showed positive reaction for RA test and LE factor and positive LE cell, and complained of fever, arthralgia and dry cough. These phenomenon disappeared after the cessation of IFN therapy.

Adult↗
PubMed1994

[Diagnostic difficulties in systemic lupus erythematosus].

A case of SLE in female 43 years old is presented. The patient was hospitalized with diagnosis of pneumonia. The correct diagnosis was made after LE-cell and antinuclear antibodies in blood serum were found. After the treatment with small prednisone doses the long years remission was given. In discussion the diagnosis difficulties and basis diagnosis are presented.

Adult↗
PubMed1995

[Collagen disease. Autoimmune disease].

Collagen disease is systemic autoimmune disease and consists of a lot of diseases with each clinical entity. for exact diagnosis, it is important to choose essential laboratory tests for the patient suspected of collagen disease in daily primary medical care. A guideline for the use of clinical laboratory tests for patients with collagen disease was proposed by the Japan Society of Clinical Pathology. This guideline was discussed repeatedly by subcommittee members of "the uses of clinical laboratory tests in daily primary medical care" and published on September of 1990. When the clinicians are suspected of the collagen disease from detailed history taking and physical examination, they must precisely interpret results of the essential laboratory tests. Urinalysis, hematology, ESR and CRP and Biochemistry show characteristic findings in the collagen disease, respectively. If further suspicion of the collagen disease is intensive, the clinicians proceed with the primary screening tests for collagen disease; rheumatoid factor, ANF, anti DNA antibody, LE test, STS and CH50. Finally, specific tests for each collagen disease are carried out to define the diagnosis; e.g. LE cell, anti-Sm antibody, IC, Coombs test and biopsy of kidney for SLE. This paper is presented on the intention of the guideline of clinical laboratory tests for the collagen disease and its issues. As it passed 4 years after published, this guideline should be more discussed and revised.

Autoimmune Diseases↗
PubMed1994

Lupus nephritis in males: 8-year experience at Siriraj Hospital.

During 1984 to 1991, 54 out of 569 lupus nephritis patients at Siriraj Hospital were male (F:M sex ratio = 10:1). Mean age of the males was 29.8 +/- 14.6 years, range 12 to 69. The three most common extrarenal manifestations were anemia, cutaneous, and musculoskeletal involvement (74.5, 51.1, and 43.9%, respectively). The major renal manifestations were edema (75.9%) with heavy proteinuria over 3.5 g/day in 62.2% and nephrotic/nephritic findings in 51.9% of cases. Hypertension was found in 35.2%. Mean serum creatinine was 2.0 +/- 1.4 mg/dl while 60.5% of cases had creatinine clearance below 50 ml/minute. Mean serum albumin was 2.6 +/- 0.8 g/dl, cholesterol 262.8 +/- 129.5 and triglycerides 343.2 +/- 244.6 mg/dl. Interestingly, hypercholesterolemia (> 250 mg/dl) was found only in 44.8% of cases with nephrotic syndrome. Antinuclear antibody was demonstrated in 91.5%, anti-dDNA antibody in 64.4% and LE cells in 40.4% of cases. Renal biopsy was done in 45 patients and 30 cases (66.7%) were classified as diffuse proliferative nephritis (WHO type IV), 15.6% of type II, 6.7% each of type III and V, with the rest of type V plus IV (4.4%). Tubulointerstitial inflammation was found in 77.3% of cases. During the follow-up period (42 +/- 35.8 months), 6 patients died. The cause of death were uremia in 3, infection in 2, and cardiac failure in 1. By life-table analysis, the probabilities of survival for 1 and 5 years were 89.5 and 80.6%, respectively. In comparison between sexes, except for a higher amount of urinary protein excretion (4.5 +/- 3.1 vs 3.5 +/- 3.0 g/day, p < 0.05), there were no statistically significant differences in clinical and pathological parameters, and probability of survival.

Adolescent↗
PubMed1995

Lupus in Chinese male: a retrospective study of 61 patients.

BACKGROUND: Systemic lupus erythematosus (SLE) has traditionally been considered a disease of women, and is uncommon in men. In recent years, several large clinical series of male lupus patients have been reported. As no known data are available for lupus in males from Taiwan, a retrospective analysis of data from male lupus patients was done to determine whether these patients differed from other series of male or female SLE patients in the literature. METHODS: Sixty-one male lupus patients, diagnosed and followed in Tri-Service General Hospital, between 1983 and 1993, were studied and their data analyzed, retrospectively. RESULTS: The mean age of diagnosis was 30 +/- 17 (mean +/- SD, range: 13-81) years. The peak age of diagnosis was between 13 and 40 years. The mean duration of follow-up was 36 +/- 36 (range: 2-256) months. The 1-, 5- and 10-year survival rates were 84%, 76% and 75%, respectively. The frequency of clinical manifestations were renal disease, 75%; malar rash, 70%; arthritis, 60%; fever, 56%; photosensitivity, 48%; pleuritis, 39%; pericarditis, 31%; alopecia, 31%; mucosal ulcers, 29%; neuropsychiatric disease, 26%; discoid lupus, 21%; vasculitis, 15%; Raynaud's phenomenon, 10%; and lymphadenopathy, 2%. The frequency of abnormal laboratory findings were antinuclear antibodies (ANA), 95%; hypocomplementemia, 77%; antibodies to double-stranded DNA (anti-dsDNA), 57%; leukopenia, 44%; lupus erythematosus (LE) cells, 39%; anti-Ro, 39%; anti-Smith antibodies (anti-Sm), 19%; thrombocytopenia, 18%; rheumatoid factor, 17%; anti-ribonucleoprotein antibody (anti-RNP), 14%; autoimmune hemolytic anemia, 8%; false-positive venereal disease research laboratory test (VDRL), 6% and anti-La, 4%. CONCLUSIONS: In a review of the 61 ethnic Chinese male lupus patients, a higher frequency of renal disease, malar rash and photosensitivity, but a lower frequency of arthritis and lymphadenopathy, compared to previous reports of Caucasians. There were no significant immunological differences from other series of male lupus, except a lower frequency of anti-dsDNA. In general, poor prognosis was noted for male lupus patients here.

Adolescent↗
PubMed1994

[A case of progressive hemifacial atrophy associated with immunological abnormalities].

We reported a case of progressive hemifacial atrophy associated with immunological abnormalities. This patient, a 35-year-old woman, had a history of high fever of unknown etiology associated with anemia and leukocytopenia at the age of 19. Eight months later, right hemifacial atrophy, alopecia and partial lipodystrophy of the right upper arm were developed. And the progression had stopped in the latter twenties. At the age of 35, she visited our hospital because of the hemifacial atrophy. Physical examination revealed right hemifacial atrophy, large alopecia at parietal and right temporal region, and partial lipodystrophy of the right upper arm. CT, MRI and ultrasound tomography of the head and the arm showed skin and scalp atrophy, temporal muscle atrophy and decrease of subcutaneous soft tissues. There was no autonomic dysfunction. Serum IgG was increased, and RA factor, anti-nuclear antibody, anti-DNA antibody and LE cell were positive. In this case, immunological abnormalities were considered to be related to the pathogenesis of hemifacial atrophy.

Adult↗
PubMed1994

Antineutrophil cytoplasmic autoantibodies in 6 patients with renal failure and systemic sclerosis.

OBJECTIVE: To elucidate the distribution and frequency of antineutrophil cytoplasmic autoantibodies (ANCA) in systemic sclerosis. METHODS: Patient sera were analyzed by indirect immunofluorescence and by enzyme linked immunosorbent assay. RESULTS: High titers for perinuclear ANCA correlated with rapid deterioration of renal function. Patients were normotensive with normal plasma renin levels, but anemia, pulmonary hemorrhage and thrombocytopenia were problematic. All patients had anti-myeloperoxidase antibodies and some had anti-nRNP, anti-DNA, anti-Sm and LE cell antibodies. Outcome was poor except for one patient treated by plasmapheresis and cyclophosphamide. CONCLUSION: A renal specific subset of systemic sclerosis is suggested.

Acute Kidney Injury↗
PubMed1994

[Nephrotic syndrome due to membranous glomerulopathy in hypocomplementemic urticarial vasculitis syndrome;--a case report].

We report a case of hypocomplementemic urticarial vasculitis syndrome (HUVS) with membranous glomerulopathy in a 62-year-old man who had a 2-month history of secondary iritis. He was transferred to our hospital because of uncontrollable edema and respiratory dysfunction. Physical examination revealed anasarca, pulmonary edema, hypertension and urticaria-like eruption on his arms. Urinalysis, blood chemistry and serological studies showed massive proteinuria (10.5g/day) with numerous granular casts, hypoalbuminemia (1.5g/dl), renal dysfunction (creatinine; 1.6mg/dl, BUN; 86mg/dl), hypercholesterolemia (total cholesterol; 455mg/dl), positive results for antinuclear factor, microsome test, thyroid test, lupus anticoaglant, antithyroglobulin test and rheumatoid factor, but LE cell or double-strand anti DNA antibody was negative. Serum complement levels were persistently low as CH50 of 13 U/ml and Clq of 6.0 micrograms/dl. The patient serum precipitated with normal human Clq by immunodiffusion analysis, indicating the presence of anti-Clq antibody. Renal biopsy revealed membranous glomerulopathy with prominent fine granular deposition of Clq along the glomerular basement membrane by immunofluorescent study and subepithelial dense deposit by electron microscopy. Corticosteroid treatment was ineffective for hypocomplementemia and nephrotic syndrome. Acute subendocardial infarction occurred on the 25th hospital day and he died of acute respiratory distress syndrome on the 45th hospital day. Autopsy revealed leucocytoclastic vasculitis in the alveolar wall. HUVS was confirmed by clinical symptoms, such as iritis and urticaria-like eruption, serum anti-Clq antibody, the absence of any specific autoantibody for systemic lupus erythematosus (SLE) and leucocytoclastic vasculitis in the alveolar wall.(ABSTRACT TRUNCATED AT 250 WORDS)

Autoantibodies↗
PubMed1994

Autoimmune myelofibrosis. A steroid-responsive cause of bone marrow fibrosis associated with systemic lupus erythematosus.

Autoimmune myelofibrosis is an uncommon disorder in which patients present with anemia and thrombocytopenia in conjunction with limited clinical manifestations of autoimmune disease or an exacerbation of previously established SLE. The presence of leukoerythroblastosis in a patient with SLE may suggest the presence of myelofibrosis. Conversely, the absence of splenomegaly in a patient with presumed idiopathic myelofibrosis may suggest an autoimmune etiology. Patients with autoimmune myelofibrosis universally have a positive ANA test and frequently have either elevated anti-DNA titers or a positive LE cell preparation. Because physical manifestations of autoimmune disease may not be evident at presentation, all patients found to have myelofibrosis should have an ANA test. Peripheral blood cytopenias in autoimmune myelofibrosis frequently respond to glucocorticoids but regression of bone marrow fibrosis may be incomplete. Hematologic response to treatment parallels that of the associated autoimmune disease.

Adolescent↗
PubMed1993

[Seropositive polyarthritis as a presentation form of angioimmunoblastic lymphadenopathy].

We present a 59-year-old male who was admitted due to fever and generalized lymphadenopathy. The patient had polyclonal hypergammaglobulinemia, Coombs-positive anemia, positive rheumatoid factor (latex 1:1280-SCAT 1:128), hypocomplementemia, negative LE cells and FAN negative. He had a 2 months history of a rheumatoid arthritis-like polyarthritis with poor response to non-steroid antiinflammatory drugs. On physical examination a mild symmetrical polyarthritis of small and large joints was seen. A lymph node biopsy showed architectural effacement, absence of germinal centers, arborization of postcapillary venules and a polymorphonuclear infiltrate that included immunoblasts. Thus, this patient fulfills the morphologic criteria of angioimmunoblastic lymphadenopathy (AILD) (Fig. 1 and 2). Our purpose was to describe the association of seropositive polyarthritis with AILD as a presentation sign. Whether this represents a case of a rheumatoid arthritis with AILD or the polyarthritis which has been described as part of the clinical picture or AILD is difficult to say due to the short time evolution of the disease.

Arthritis↗
PubMed1993

A case complicated with SLE during maintenance hemodialysis.

A 40-year-old woman suffered from toxemia of pregnancy in 1977 and was admitted to hospital. Thereafter, she developed nephrotic syndrome, underwent a renal biopsy, and a diagnosis of membranoproliferative glomerulonephritis (MPGN) was made. She received steroid therapy, immunosuppressive drug and anticoagulant therapy, and recovered sufficiently to be discharged from hospital in April, 1979. During subsequent ambulatory treatment at our outpatient department, her renal function deteriorated gradually, and maintenance hemodialysis was started from June, 1990. In July, 1991, she was admitted to our hospital with pleurisy and pericarditis. There was no improvement despite antibiotic treatments. Laboratory data revealed leukopenia and lymphopenia. Under suspicion of systemic lupus erythematosus (SLE), relevant tests were carried out. Immunological abnormalities such as positive LE cells and the presence of various autoantibodies, together with clinical signs of hypersensitivity to sunlight, stomatitis and serositis, satisfied the diagnostic criteria of the ARA and a diagnosis of SLE was made. This case did not exhibit any clinical or serological abnormalities except for the renal disorder for a 10-year period after the histological diagnosis of MPGN, but was eventually diagnosed as SLE as a result of the manifestation of SLE symptoms for the first time after one year of maintenance hemodialysis. Immunological abnormalities and SLE during maintenance hemodialysis are discussed in relation to other reports.

Adult↗
PubMed1993

[Enzyme-immunoassay using recombinant antigen].

Since Hargraves et al reported the LE cells in 1948, many antinuclear antibodies (ANAs) have been classified using various immunological methods, and have become an important tool for the diagnosis of various connective tissue diseases. Although the enzyme-immunoassay (EIA) is known as a safe and sensitive quantitative-method to detect ANAs, it has not been used routinely because of difficulty to prepare a large amount of antigens reactive with autoantibodies specifically. Many investigators have established EIA systems using recombinant proteins to overcome this problem and some of them have been useful for the diagnosis of connective tissue diseases showing highly specific and sensitive reactions to autoantibodies. However, some sera positive for autoantibodies to several nuclear antigens such as U1 RNP, Sm and SS-A, do not react with the recombinant proteins because those autoantibodies often recognize the conformational profiles of the autoantigens. Although many problems still remain to be solved, EIA using recombinant protein is superior as a quantitative method than other methods such as double immunodiffusion (DID), and it can provide much more useful information to estimate the effect of treatment. Therefore, at this moment, the use of both DID and EIA is recommended to diagnose the patients and to evaluate the disease activities, and may clarify the clinical significance of EIA using recombinant antigens.

Antibodies, Antinuclear↗
PubMed1996

[Cardiac tamponade disclosing systemic lupus erythematosus].

Cardiac tamponade secondary to systemic lupus erythematosus is rare and has a very serious prognosis. The authors report a case of cardiac tamponade confirmed by echocardiography, which constituted the presenting sign of systemic lupus erythematosus in a 20-year-old patient, who required emergency pericardial aspiration. The diagnosis of systemic lupus erythematosus was established on the basis of the combination of pericardial involvement, non-erosive arthritis, leukopenia with lymphopenia, presence of LE cells and anti-native DNA antibodies and positive antinuclear antibody titre of 1/2560. The clinical course was favourable in response to 3 months of corticosteroid treatment. The possibility of SLE should be considered in any case of cardiac tamponade in a young patient in which the aetiology is not explained.

Adult↗
PubMed1996

Clinical features of patients with juvenile onset mixed connective tissue disease: analysis of data collected in a nationwide collaborative study in Japan.

OBJECTIVE: To analyze the clinical features and outcome of juvenile-onset mixed connective tissue disease (MCTD). METHODS: Clinical and laboratory findings were compared in 2 groups of MCTD patients divided according to age at onset: juvenile onset: under 16 yrs: adult onset: 16 yrs or older). RESULTS: Systemic lupus erythematosus-like symptoms, such as facial erythema, photosensitivity. LE cells, lymphadenopathy, and cellular casts, were more frequent in juvenile onset MCTD than in the adult form of the disease. On the other hand, scleroderma-like symptoms, such as esophageal hypomotility, scleroderma-like lesions evident on skin biopsy, pulmonary involvement, proximal scleroderma, and pitting scars, were less frequent in juvenile onset MCTD than in the adult form. Patients with juvenile onset MCTD more frequently met the classification criteria for systemic lupus erythematosus (SLE) and less frequently met those for progressive systemic sclerosis (SSc), compared to patients with adult onset MCTD. At disease onset, hand edema and stiffness were observed less frequently in juvenile onset MCTD than in the adult form. Furthermore, the mortality rate was lower in the former than in the latter (2.8% vs 8.4%). CONCLUSION: Although previous studies have reported severe symptoms and adverse outcome for juvenile onset MCTD, we conclude from this nationwide study in Japan that patients with juvenile onset MCTD exhibit more SLE-like and fewer SSc-like features and have a relatively favorable outcome.

Adolescent↗
PubMedDate not supplied

Haematological changes in hyperimmunized rabbits treated with hydrazinophthalazines for long time periods.

Haematological investigations of peripheral blood and bone marrow were performed in rabbits receiving hydrazinophthalazines (hydralazine or binazin) with or without simultaneous hyperimmunization by means of repeated injections of bovine albumin. It was found that long-term administration of hydralazine caused appearance of LE factor in the serum and LE cells in blood. Less evident changes were caused by Binazin administration. Hyperimmunization caused weaknening of the effects of hydralazinophthalazines. Haematological investigations demonstrated hypochromic anaemia.

Animals↗
PubMedDate not supplied

[Dermatopolymyositis and overlap syndromes].

Clinical characteristics and immunoserological features of patients with overlap myositis treated in Clinic for Rheumatology of Military Medical Academy in the period 1978-1996 are presented in the study. Retrospective and partially prospective study included 16 patients with the diagnosis of dermatopolymyositis associated with some other systemic disease of connective tissue (SDCT) in overlap syndrome. There were 12-females of average age 37.33 years and 4 males of average age 52.25 years. Complete laboratory investigation included the determination of muscular enzymes values. Immunoserological analyses were also performed (RF, Le cells, ANA, ICs, immunoglobulins, cryoglobulins and complement), EMNG examination of pelvis and shoulder and pH examination of skin, subcutaneous tissue and muscles. The combination of the diseases was found in 11 patients, 7 were with PSS, 2 with SLE, 1 with RA and one with PAN. Three diseases were associated in 4 patients, SLE and RA in 2, PSS and RA in 1, thyroiditis and sicca complex in 1. Five-fold combination of disease was found in 1 patient. More frequent occurrence of fever, polyarthritis, skin manifestations, Raynoud's phenomenon, the involvement of serosas, lungs and heart, histological finding of skin and/or muscle vasculitis and larger presence of ANA, ICs, hypergammaglobulinemia and hypocomplementemia pointed out the dominant disorder of humoral immunity which was most probably connected with associated SDCT.

Adult↗