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PubMed1992

Systemic lupus erythematosus in men: clinical and immunological characteristics.

Although systemic lupus erythematosus (SLE) has traditionally been considered a disease of women, men may also be affected. Thirty of 261 patients (12%) with SLE seen in this hospital were men. Arthritis was less common as a first symptom in the men, although this group of patients had discoid lesions and serositis more often than the women. During the follow up a lower incidence of arthritis and malar rash and a higher incidence of other skin complications including discoid lesions and subcutaneous lupus erythematosus was found in the men. The incidence of nephropathy, neurological disease, thrombocytopenia, vasculitis, and serositis, was similar in the two groups. No significant immunological differences were found between men and women. These features indicate that several gender associated clinical differences may be present in patients with SLE.

Adolescent↗
PubMed1994

The morbidity and mortality associated with childhood onset systemic lupus erythematosus.

From 1977 till 1991, the diagnosis of systemic lupus erythematosus was made on 137 children aged 18 years or under in Chang Gung Memorial Hospital. The medical records were reviewed and the clinical data were analysed with emphasis on the morbidity and mortality of this disease. The clinical and laboratory characteristics were similar to the findings from most other reports. Renal failure accounted for 8% of the initial presentation. The non-infectious complications were, in the order of frequencies, hypertension, avascular necrosis of femoral head, cataract, encephalopathy, and venous thrombosis. Sepsis, cutaneous infection and urinary tract infection were the frequently encountered infectious complications. The major causes of death in childhood onset systemic lupus erythematosus were sepsis (42%) and renal failure (30.7%). Forty patients were lost to follow-up. The 5-year survival rate, calculated by life-table, was 76.3%.

Adolescent↗
PubMed1985

The value of complement and immune complex determinations in monitoring disease activity in patients with systemic lupus erythematosus.

Serial serum samples from 33 patients with systemic lupus erythematosus were tested for CH50, C3, C4, and circulating immune complexes. Circulating immune complexes were determined by 5 different assays. Disease activity was determined by a scoring system we devised. On an interpatient analysis, overall disease activity correlated significantly with levels of CH50, C3, and a positive result on C1q binding assay. However, with the exception of depressed C3 levels, the sensitivity, specificity, and predictive values of all the serologic tests were low. An intrapatient analysis revealed a patient-specific activity parameter in 11 of the 33 patients. We conclude that these serologic tests are not reliable in assessing disease activity in patients with systemic lupus erythematosus, although a small subgroup of patients will exhibit a patient-specific activity parameter.

Adolescent↗
PubMed1997

Discoid lupus erythematosus presenting as asymmetric posterior blepharitis.

PURPOSE: To describe the ophthalmic findings of patients with discoid lupus erythematosus. METHOD: We describe two women who originally were thought to have asymmetric posterior blepharitis; however, the involved eyelid also had an erythematous, scaly cutaneous lesion. RESULT: In both patients, histology and immunofluorescence studies performed on cutaneous biopsy specimens established the diagnosis of discoid lupus erythematosus. CONCLUSIONS: It is important to diagnose discoid lupus of the eyelids because misdiagnosis can delay treatment and thus lead to deformities of the eyelid margin. Misdiagnosis can also lead to a complicated full-thickness eyelid biopsy and delay the diagnosis of systemic lupus erythematosus.

Adult↗
PubMed2004

Systemic lupus erythematosus trials: successes and issues.

PURPOSE OF REVIEW: The purpose of this review is to discuss the most recent published clinical trials for systemic lupus erythematosus and to identify important issues that have arisen in association with the search for new therapies for systemic lupus erythematosus, as well as new regimens or indications for the use of "standard-of-care" agents such as corticosteroids and cyclophosphamide. RECENT FINDINGS: Important developments have occurred during the past 2 years as interest in this area has increased, largely because of the participation of pharmaceutical and biotechnical companies in the development and testing of novel agents for systemic lupus erythematosus. Several important large-scale, multicenter, randomized controlled trials have been completed, but none has yet resulted in a new, approved indication for systemic lupus erythematosus. Many issues in the identification of new therapeutic modalities remain. These include the fact that a majority of published reports include either small numbers of patients in controlled trials that lack statistical power to draw conclusions, or are uncontrolled anecdotal series or individual case reports. Among the larger controlled trials, a pervasive issue in the failure to reach statistical significance may be the initial study design. Inclusion of patients with mild and/or stable disease activity does not allow for an effect size sufficient to show differences in treatment arms without recruitment of very large numbers of subjects. Finally, several potentially important trials have been reported only in abstract form to date. Further assessment of the results must await formal publication of these studies.

Biological Products↗
PubMed1999

Lupus erythematosus associated with erythema multiforme: does Rowell's syndrome exist?

We describe a patient with lupus erythematosus who experienced an unusual erythema multiforme-like eruption suggestive of Rowell's syndrome. We compare our case and 9 other reports of lupus erythematosus associated with erythema multiforme to the 4 cases reported by Rowell. Our findings indicate that Rowell's original criteria are not well preserved. The coexistence of lupus erythematosus with erythema multiforme does not impart any unusual characteristics to either disease, and the immunologic disturbances in such patients are probably coincidental.

Adult↗
PubMed2000

[Low-flow priapism associated with systemic lupus erythematosus and nephrotic syndrome].

OBJECTIVE: To present a case of priapism associated with systemic lupus erythematosus and nephrotic syndrome. METHODS: A 29-year-old male patient with a history of multiple pathologies consulted at the emergency services with painful priapism of 12 hours' duration. The intracavernosal aspirated blood had a venous appearance. Among the risk factors analyzed, systemic lupus erythematosus and nephrotic syndrome are recognized to cause hypercoagulability, a condition that can lead to low flow priapism. RESULTS: There was no response to lavage-aspiration or intracorporeal administration of an alpha-adrenergic agonist. The high surgical risk of the patient advised against surgery. The priapism gradually diminished until complete remission on the sixth day. The patient has remained without spontaneous erection. CONCLUSION: Systemic lupus erythematosus and nephrotic syndrome can cause low flow priapism. The best treatment is by prevention with adequate antithrombotic prophylaxis.

Adult↗
PubMedDate not supplied

Valvulitis involving a bioprosthetic valve in a patient with systemic lupus erythematosus.

A 37-year-old man with systemic lupus erythematosus, who underwent an aortic valve replacement with a Carpentier-Edwards porcine valve for severe aortic insufficiency, was admitted to the hospital with pulmonary edema. Transesophageal echocardiography revealed severe aortic insufficiency arising from partial dehiscence of the valve sewing ring, as well as centrally from the valve cusp. In addition, marked thickening of the mitral valve was observed with severe eccentric regurgitation. At surgery, valvulitis of the native mitral and bioprosthetic aortic valves was demonstrated, with a perforation of the porcine valve cusp. After replacement of both valves, the patient had a stormy postoperative course with recurrent communications between the left ventricle and atrium requiring multiple surgeries and eventually died. This case illustrates the severity of valvulopathy and ensuing complications that can affect patients with systemic lupus erythematosus and demonstrates that the valvulopathy can affect bioprosthetic valves, a finding that has significant implications as to the type of valve replacement in these patients.

Adult↗
PubMed1996

Systemic lupus erythematosus with erythema multiforme-like lesions and histiocytic necrotizing lymphadenitis--a case report.

A 9-year-old Chinese girl with systemic lupus erythematosus presented initially with persistent cervical lymphadenopathy without any constitutional symptoms. The histology of the involved lymph node showed histiocytic necrotizing lymphadenitis and the initial clinical diagnosis of Kikuchi-Fujimoto disease was made. The patient subsequently developed fever, oral ulcerations, epistaxis, generalised myalgia and erythema multiforme-like skin lesions over the face and upper arms. Investigations showed anaemia, leucopenia, hypocomplementemia, a positive antinuclear antibody and a positive anti-double-stranded DNA antibody test. The clinical diagnosis was revised to systemic lupus erythematosus. The histological picture of histiocytic necrotizing lymphadenitis can occur in systemic lupus erythematosus and may be indistinguishable from that seen in Kikuchi-Fujimoto disease. The diagnosis of Kikuchi-Fujimoto disease should be confined to a benign clinical subset of histiocytic necrotizing lymphadenitis which resolves spontaneously without treatment. The occurrence of erythema multiforme-like lesions in lupus erythematosus is unusual and together with the characteristic serologic abnormalities, fits Rowell's syndrome.

Child↗
PubMed1991

NIH conference. Systemic lupus erythematosus.

Although the cause of systemic lupus erythematosus remains unknown, pathogenic mechanisms are becoming clearer. Both genetic and environmental factors have been implicated in the induction and in the perpetuation of lupus. Implicated environmental triggers include ultraviolet light, chemicals (hydrazines, hair dyes, drugs), some foods, and possibly infectious agents. Lupus is mediated by the immune system. Patients have excess numbers of antibody-forming cells, including those that produce antibodies reactive with self-antigens. Patients also have an increased number of activated T cells, some of which help B cells to produce autoantibodies. A loss of tolerance is a critical immune abnormality in lupus; many of the activated helper T cells may result from a failure in normal tolerance mechanisms. A hematopoietic stem-cell defect could give rise to both B- and T-cell abnormalities. Such a stem-cell abnormality might lead to both a loss of self-tolerance and polyclonal B-cell activation. Antigen-driven, T-cell-dependent expansion of B-cell clones would then give rise to pathogenic autoantibodies, including anti-DNA. We believe that lupus is a syndrome: Patients differ regarding specific inciting factors and immune defects. Some patients have genetically conditioned abnormalities, similar to those found in mice with lupus; others have a combination of genetic and acquired defects. We hope that insights into pathogenesis lead to improved and more individualized therapy.

Animals↗
PubMedDate not supplied

Childhood discoid lupus erythematosus: a Tunisian retrospective study of 16 cases.

Discoid lupus erythematosus (DLE) is uncommon in children. The clinical features of childhood DLE are similar to those of adult DLE in presentation and chronic course. However, children have a particularly high level of transition to systemic disease. We undertook a retrospective study of 16 children with DLE ranging in age from 2 to 15 years, seen over a 9-year period. Six were less than 10 years old at the onset of the disease. The sex ratio was equal. The frequency of childhood DLE was about 7% of the total number of DLE patients seen in our department. Photosensitivity was defined as a clinical history of induction or exacerbation of discoid lesions following sun exposure, and was present in 81% of patients. There was no progression to systemic lupus erythematosus (SLE); an average follow-up time was 10.5 months (2-30 months). We would like to emphasize the increased frequency of childhood DLE in our country and the importance of photosensitivity. However, follow-up data regarding transition to SLE is lacking, therefore we are unable to offer a prognosis to our patients.

Adolescent↗
PubMedDate not supplied

Mucinous cystadenoma of the appendix in a patient with systemic lupus erythematosus.

A 38-year-old female with systemic lupus erythematosus presented with abdominal pain, diarrhea and iron-deficient anemia. Computed tomogram showed a 2 x 4 cm inhomogeneous lesion of the right adnexa. An unusual mass was identified extending from the appendiceal orifice at colonoscopy, and an 8 cm tubular appendix, apparently prolapsed into the cecum, was identified at celiotomy. An appendectomy with cecectomy was performed. On cut section, mucin was extruded from the lumen of the appendix. A mucinous neoplasm of the appendix with mucinous dissection to the serosal surface was reported at the time of frozen section. No gross ovarian pathology or peritoneal implants were noted. Cystadenoma with associated mucocele formation was verified by permanent histology. Mucocele of the vermiform appendix is a rare condition associated with neoplastic transformation in approximately 75% of all cases. Benign mucinous cystadenoma of the appendix should be differentiated from cystadenocarcinoma by frozen section at the time of celiotomy to ensure appropriate treatment. While systemic lupus erythematosus can lead to cutaneous mucinosis, an association with mucinous cystadenoma of the appendix has not been previously reported. Surveillance for metachronous colonic neoplasms is warranted in patients diagnosed with a mucinous neoplasm of the appendix.

Adult↗
PubMed1989

Dyslipoproteinemia in murine systemic lupus erythematosus.

Dyslipoproteinemia, a feature of systemic lupus erythematosus may contribute to premature atherosclerosis. In order to develop an experimental model for this dyslipoproteinemia we measured plasma concentrations of lipoproteins in juvenile NZB/W (lupus) and NZB/B (control) mice. Additionally to evaluate the effects of a diet rich in n - 3 fatty acids we measured lipoprotein concentrations in mice on normal or menhaden oil-enriched diets. The lupus mice had elevated triglycerides compared to the controls, similar to that seen in human SLE patients (161 +/- 31 vs 113 +/- 13 mg/dl, P less than 0.003). In contrast, the menhaden oil diet fed NZB/W mice had triglycerides similar to the NZB/B control fed group. In the NZB/W murine SLE model, dyslipoproteinemia is an early sign of disease as has been shown in man, therefore this model will be useful in elucidating the mechanism of dyslipoproteinemia in SLE.

Animals↗
PubMed1989

[A case of systemic lupus erythematosus with neurological manifestations as initial symptoms].

According to the past reports, neuropsychiatric manifestations have been seen in 10-75% of patients with systemic lupus erythematosus and are second only to renal involvement as a cause of death. The clinical feature is multiple. And cerebrovascular diseases due to systemic lupus erythematosus are detected in 3-16% of the neuropsychiatric manifestations. Occlusion of the intracranial major arteries is less frequently found in other cerebrovascular diseases. And central nervous system involvement usually occurs at some intermediate or terminal stage of systemic lupus erythematosus, so is rarely regarded as one of the initial symptoms. We studied the case of a patient with systemic lupus erythematosus with occlusion of the right middle cerebral artery indicated by angitis and 'string of beads' appearance of the right internal carotid artery indicated by fibromuscular dysplasia. The patient was a 38 year old female and began to feel weakness in the left hand and developed mild-left hemiparesis due to infarction of right temporo-parieto-occipital lesion which was revealed by CT scan. Carotid angiograph showed irregularity at the right middle cerebral artery and 'string of beads' appearance of the right internal carotid artery. Gradually neurological manifestations improved, but a facial 'butterfly' rash, palmar erythema and polyarthritis were detected.(ABSTRACT TRUNCATED AT 250 WORDS)

Adult↗
PubMed1991

Detection of anti-basement membrane zone antibodies in bullous systemic lupus erythematosus.

We describe a 42-year-old black woman with long-standing systemic lupus erythematosus in whom vesiculobullous lesions developed. Routine histologic and immunologic studies fulfilled the criteria for the diagnosis of bullous systemic lupus erythematosus. Indirect immunofluorescence showed antinuclear antibodies without basement membrane zone fluorescence. We destroyed the nuclear antigens of the indirect immunofluorescence substrate with 2 mol/L sodium chloride, which unmasked basement membrane zone linear IgG staining. We also confirmed anti-basement membrane zone antibodies by employing a new technique of direct immunofluorescence on sodium chloride-split skin. Our finding prove that a thorough search for anti-basement membrane zone antibodies can be revealing. Our results support the idea that a subset of bullous systemic lupus erythematosus has the staining characteristics of epidermolysis acquisita, with the dermal side of the split skin showing linear immunoglobulin deposition.

Adult↗
PubMed1990

Stroke in systemic lupus erythematosus.

We investigated the clinical and pathologic characteristics of stroke in 234 patients with systemic lupus erythematosus. Thirteen patients (5.6%) developed cerebrovascular disease. Cerebral infarction was noted in eight, cerebral hemorrhage in two, and subarachnoid hemorrhage in three. In seven (54%) of these 13 patients, stroke occurred less than or equal to 5 years after systemic lupus erythematosus was diagnosed. Among the predisposing risk factors for stroke, hypertension was the most important. Lupus anticoagulant was detected in three (38%) and anticardiolipin antibody in three (43% of seven investigated) of the patients with infarction. Evaluation of the clinical manifestations and autoantibodies indicated that renal involvement and high titers of anti-deoxyribonucleic acid antibody were more frequent in the stroke group than in the non-stroke group. Autopsy studies on six of the patients with stroke revealed small infarcts and hemorrhages in all, but in no case was true angiitis observed. Libman-Sacks endocarditis was found in two of the three patients with infarction. In conclusion, the important contributory factor to the development of stroke in patients with systemic lupus erythematosus is considered to be hypertension mediated by immunologic abnormalities. Antiphospholipid antibodies and Libman-Sacks endocarditis are closely associated with occlusive cerebrovascular disease.

Adolescent↗
PubMed1988

Danazol for the treatment of refractory autoimmune thrombocytopenia in systemic lupus erythematosus.

STUDY OBJECTIVE: to determine the efficacy of danazol therapy in patients with systemic lupus erythematosus with severe autoimmune thrombocytopenia refractory to other therapies. DESIGN: noncontrolled clinical trial, with a minimum of 8 weeks of therapy, the maximum determined by clinical response. SETTING: referral-based rheumatology clinic at an army medical center. PATIENTS: sequential sample of six patients with systemic lupus erythematosus with severe autoimmune thrombocytopenia refractory to high-dose glucocorticoids. Four patients also failed splenectomy, or cytotoxic drugs, or both. INTERVENTIONS: danazol, 200 mg four times per day, was added to the previous therapeutic regimen for at least 2 months. MEASUREMENTS AND MAIN RESULTS: all six patients had normal platelet counts within 6 weeks of starting danazol treatment. After resolution of thrombocytopenia for at least 1 month, immunosuppressive medications were tapered; one patient had a relapse. During an average follow-up of 12 months, the danazol dose was lowered in the five remaining patients but could not be discontinued without recurrence of thrombocytopenia. During danazol therapy, platelet-bound IgG antibodies and circulating immune complexes did not decrease significantly. Danazol was well tolerated. CONCLUSIONS: danazol appears to be a useful adjunctive treatment for refractory autoimmune thrombocytopenia associated with systemic lupus erythematosus.

Adult↗
PubMed1997

[Coronary involvement in systemic lupus erythematosus].

BACKGROUND: Coronary artery disease is an uncommon event in lupus erythematosus. The mechanisms responsible for coronary occlusion are probably complex and intermixed. We report three patients with lupus erythematosus and antiphospholipid antibodies who had coronary artery disease diagnosed with coronary angiogram. OBSERVATION: Coronary artery disease occurred in three young patients aged from 21 to 35 years 3 to 11 years after the onset of lupus. They all had antiphospholipid antibodies. They had been treated with corticosteroids for 6 to 36 months. Two of them were smokers. Angiograms showed coronary occlusion two patients while the third one had probable myocardial microvasculopathy. The lupus was quiescent in all cases when coronary artery disease occurred. DISCUSSION: Antiphospholipid antibodies associated with smoking may be involved in the pathogenesis of coronary artery disease in these 3 patients.

Adrenal Cortex Hormones↗