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PubMed2001

Molecular genetics of cutaneous lupus erythematosus.

The cutaneous forms of lupus erythematosus (LE) are true complex traits, susceptibility to which is determined by multiple factors. Good evidence exists for both genetic and environmental components to this complexity. Several different experimental techniques have found the strongest genetic associations with cutaneous LE to include sequence polymorphisms of genes encoding HLA, TNF-alpha and complement molecules, particularly in anti-Ro-positive patients. Abnormal expression of multiple other cytokines, adhesion molecules and cellular proteins (such as Ro and La) points towards a range of candidate genes that are currently being examined in cutaneous LE. Combinations of specific polymorphisms of genes encoding these immunoregulatory molecules may determine individual susceptibility to LE.

Cytokines↗
PubMed1991

Bullous lesions in cutaneous lupus erythematosus.

Seventeen patients with cutaneous lupus erythematosus (LE) were observed to have bulla formation. Their clinical, histopathologic, and immunopathologic findings are reported. All biopsy specimens studied revealed moderate to severe vacuolating degeneration of the basal cell layer as well as lymphocytic inflammatory infiltrate involving the dermoepidermal junction. Immunoglobulins or C3 (or both) was deposited in the basement membrane zone in eight of nine patients in whom direct immunofluorescence study was performed. We believe that the immune reaction in the papillary dermis and the severe vacuolating degeneration in the basal cell layer result in the separation of epidermis and dermis, and thus bullous formation, in some patients with LE. This phenomenon can be observed not only in systemic LE but also in subacute cutaneous LE and discoid LE.

Adult↗
PubMedDate not supplied

A case of lichen planus-lupus erythematosus overlap syndrome with eyelid involvement.

PURPOSE: To report a case of lichen planus-lupus erythematosus overlap syndrome with eyelid involvement. Lichen planus and lupus erythematosus infrequently coexist in the same patients. Ocular involvement has rarely been reported for both diseases. CASE REPORT: We describe a case of lichen planus-lupus erythematosus overlap syndrome with eyelid involvement. Histopathologic and immunofluorescent studies were done on buccal, lip, left conjunctival, malar, auricular and scalp lesions. The immunopathologic features of the conjunctiva, buccal mucosa and lip were consistent with lichen planus, while those of the malar, auricular and scalp lesions favoured lupus erythematosus. RESULTS: The patient was successfully treated with hydroxychloroquine 200 mg/day and all lesions responded to therapy within weeks. CONCLUSIONS: This is a rare example of two coexisting autoimmune disease entities: lichen planus of the oral mucosa, lip, eyelid and discoid lupus erythematosus of the skin. To our knowledge, this is the first reported case of lichen planus-lupus erythematosus overlap syndrome with eyelid involvement.

Adult↗
PubMed2003

Treatment of lupus erythematosus with pulsed dye laser.

BACKGROUND AND OBJECTIVES: The treatment of cutaneous lupus erythematosus (CLE) with dye and argon laser has been evaluated in a number of articles in recent years. The improvement of telangiectasias and chronic erythema of the cutaneous lesions was based on the selective photothermolysis ablation of the dilated capillaries and venules. STUDY DESIGN/MATERIALS AND METHODS: We describe the results of the treatment of cutaneous lesions of 14 patients; eight with discoid lupus erythematosus (DLE) and six with systemic lupus erythematosus (SLE). Three patients received a treatment with flashlamp pulsed dye laser (FPDL) (585 nm, 450 microseconds) with fluences in the range from 5 to 7.75 J/cm(2); the other 11 patients were treated with long pulsed dye laser (LPDL) (595 nm, 1.5-10 milliseconds) with fluences in the range from 6 to 13 J/cm(2) depending on the pulse duration. RESULTS: During a median follow-up of 10 months, we observed an average improvement in over 60% of the lesions. A few side effects were observed in all patients: four had transient hyperpigmentation and one patient had light scarring. Three patients had a relapse after more than 1 year; they were then offered conventional treatment. CONCLUSIONS: We confirm that pulsed dye laser is a good alternative treatment for the erythema in active cutaneous lesions of lupus erythematosus (LE).

Adult↗
PubMed2006

A case of systemic lupus erythematosus presenting with protein-losing enteropathy.

We report an unusual case of systemic lupus erythematosus presented with protein-losing enteropathy. A 24-year-old girl was referred to our hospital with generalized edema, thrombocytopenia, hypoalbuminemia, hypercholesterolemia, hypocomplementemia, antinuclear antibody (ANA) (speckled pattern) and anti- SSA/Ro positivities, and elevated CA125 antigen appeared in the blood examination. On the radiological studies, she had mild pleural effusion and moderate ascites which were transudate. A diagnosis of protein-losing enteropathy was made on the basis of increased 99mTc-labelled human immunoglobulin scintigram showing abnormal radioactivity. Endoscopic gastric, duodenal and jejunal biopsies showed chronic inflammation, but vasculitis and immune complex deposition findings were not present. Renal biopsy revealed no definitive findings of lupus nephritis. By the administration of corticosteroids, hypoalbuminemia began to improve, but steroid doses were decreased due to steroid-induced myopathy. Temporary hemiparesis and facial paralysis developed in the patients' follow up. Her cranial magnetic resonance imaging revealed chronic ischemia, and the patient was considered to have neurological involvement due to systemic lupus erythematosus. protein-losing enteropathy and other symptoms then improved dramatically after monthly intravenous cyclophosphamide (three times) combined with oral low-dose corticosteroids. The combination of azathioprine and low-dose steroids was used as maintenance medication. Although about 30 protein-losing enteropathy -associated systemic lupus erythematosus cases have been reported, the patients having initial symptoms as protein-losing enteropathy are rare in the literature. Protein-losing enteropathy -associated systemic lupus erythematosus cases probably represent a subgroup of systemic lupus erythematosus, the characteristics of which are hypocomplementemia, protein-losing enteropathy, ANA positivity showing speckled pattern and anti-ds DNA negativities. In the patients with systemic lupus erythematosus with edema and hypoalbuminemia without renal protein loss, protein-losing enteropathy-associated systemic lupus erythematosus should be kept in mind.

Adrenal Cortex Hormones↗
PubMed2004

Coronary artery aneurysm with systemic lupus erythematosus.

Coronary artery aneurysms rarely develop in patients with systemic lupus erythematosus. We herein describe a case of a right coronary artery aneurysm associated with systemic lupus erythematosus. A 49-year-old woman with known systemic lupus erythematosus presented with an acute myocardial infarction. A coronary artery aneurysm and thrombo-occlusion in the right coronary artery necessitated percutaneous coronary intervention. She experienced three myocardial infarctions during the following two months. A coronary artery dissection occurred during the percutaneous coronary intervention at the time of the last myocardial infarction, and emergency coronary artery bypass grafting was successfully performed. Difficulties in treatment with percutaneous coronary intervention and the fact that arteritis is a possible cause of a coronary artery aneurysm may indicate that surgical therapy, including coronary artery bypass grafting with or without the obliteration of an aneurysm, is the treatment of choice for a coronary artery aneurysm with systemic lupus erythematosus.

Aneurysm↗
PubMed2004

[Lipid profile in patients with systemic lupus erythematosus, with special focus on lipoprotein(a) in lupus nephritis].

Systemic lupus erythematosus (SLE) is a multifactorial polysystemic autoimmune disorder. Although life expectance in SLE has been improved by adequate immune suppressive therapy, the importance of chronic renal failure has not been reduced. Among late complications of the disease accelerated atherosclerosis attempts increasing attention. Dyslipoproteinemia and increased concentration of lipoproteins are important risk factors of atherosclerotic cardiovascular complication in SLE. Serum lipid parameters of 50 patients with lupus were examined in the present work. Thirty patients had histologically proven lupus nephritis (LN+), while the other group did not have renal involvement (LN-). Serum triglyceride, total cholesterol, LDL-C and apolipoprotein B (apoB) concentrations were significantly higher in the lupus nephritis (LN+) group. On the other hand, HDL-C and apoAI levels were also elevated in patients with LN. As a consequence of that, LDL-C/HDL-C and the apoB/apoAI ratios did not differ between patients with or without kidney involvement. This concluded the authors to measure the concentration of lipoprotein (a) in SLE patients, as Lp(a) is known to be an independent risk factor of atherosclerosis. Results indicated a significantly increased Lp(a) concentration in patients with lupus nephritis as compared to the LN- group. All but 2 patients without kidney involvement had lower than 100 mg/L Lp(a) concentration, while 27% of patients with lupus nephritis has an Lp(a) level between 100-300 mg/L. Further more, Lp(a) concentration was higher than 300 mg/L in 13% of the LN+ group. In a good correlation of these observations patients with nephritis suffered more frequently from deep venous thrombosis and ischaemic heart disease. The frequencies of hypertension and non-insulin dependent diabetes mellitus were slightly elevated in patients with nephritis. Present results suggest the importance of elevated lipoprotein (a) concentration in patients with lupus nephritis, further increasing the risk of athero-thrombotic cardiovascular complications.

Adult↗
PubMed2002

[Neonatal lupus erythematosus and neurologic involvement: an incidental association?].

UNLABELLED: Neonatal lupus erythematosus is a rare disorder characterized by cutaneous lesions of the face and/or congenital heart block. The transplacental transfer of maternal anti-Ro/SSA, anti-La/SSB, or anti-U1RNP antibodies is responsible for the development of the disease. Few cases of neonatal lupus erythematosus with neurological involvement were reported in the medical literature. CASE REPORT: A 36-week GA female infant presented with neonatal lupus erythematosus comprising cutaneous, hematologic and hepatic disorders with a favorable outcome. However, cutaneous atrophy and hyperpigmentation persisted. Spastic paraparesis was diagnosed at the age of six months. CONCLUSION: The neurological lesions in neonatal lupus erythematosus could either be related to the presence of anti-Ro/SSA antibodies of maternal origin, or of anticardiolipin antibodies.

Antibodies, Anticardiolipin↗
PubMed1988

[Pregnancy in patients with visceral lupus erythematosus, with special reference to kidney involvement].

Of 44 female patients with lupus erythematosus visceralis after and with manifestation of the disease, respectively, in eight women ten pregnancies developed, out of them three viable births between the 33rd and 40th week of pregnancy, one stillbirth in the 37th week of pregnancy, one miscarriage in the IIIrd month of pregnancy and five interruptions of pregnancy. It is reported on the course of the disease and pregnancy of five women, taking into particular consideration the lupus nephritis. With the help of the literature and on the basis of own experiences course of the lupus erythematosus visceralis and lupus nephritis during pregnancy, the influence of the lupus erythematosus visceralis on pregnancy and development of the child, peculiarities of the treatment of lupus erythematosus visceralis during pregnancy as well as questions of prevention and interruption of pregnancy are discussed. Finally is tried to draw universal conclusions.

Adult↗
PubMed1979

Discoid lupus erythematosus in the Nigerians.

Results of a detailed clinical and laboratory study of 37 Nigerian patients with chronic discoid lupus erythematosus are presented. Patients with chronic discoid lupus erythematosus constituted 0.46% of all out-patients seen in the skin clinic between May 1974 and December 1977. A preponderance of females was noticed (female/male ratio of 5:1), while the age distribution of African patients corresponded to values characteristic for the condition seen in other geographical regions. Several morphological types of the condition have been seen. The vitiligoid variant of chronic discoid lupus erythematosus seems to be common in West Africans. Sixteen out of 37 patients presented laboratory abnormalities considered as markers of the association between chronic and systemic lupus erythematosus. Their significance, however is, uncertain as it has been demonstrated on several occasions that in a tropical milieu heavy parasitic infections produce marked immunological disturbances. The problem of the relationship between chronic and systemic lupus erythematosus is discussed and the literature on the incidence of chronic discoid lupus erythematosus in various African countries is reviewed.

Adolescent↗
PubMed1991

Disturbances in the tissue plasminogen activator/plasminogen activator inhibitor (TPA/PAI) system in systemic lupus erythematosus.

Increased thrombogenesis observed in systemic lupus erythematosus (SLE) is derived from multiple mechanisms, including: Enhanced coagulation factor VIII:VWf activity, lupus anticoagulants, anti-phospholipid antibodies, acquired deficiencies of natural anti-thrombotic mechanisms (protein C, protein S, anti-thrombin III), and impaired fibrinolytic mechanisms. We studied the fibrinolytic mechanisms of 18 patients with systemic lupus erythematosus, selected carefully to avoid other possible causes of abnormalities in the fibrinolytic activity. Despite the fact that the euglobulin lysis time in steady state was normal in all instances, disturbances in the tissue plasminogen activator/plasminogen activator inhibitor (TPA/PAI) system were found in all SLE patients: TPA activity was undetectable in all cases, whereas it was above 0.4 IU/ml in a control group. In 72 percent of patients, the undetectable TPA activity was correlated with abnormally high PAI activity; PAI levels were normal in all members of the control group, their mean value being 0.74 versus 8.63 IU/ml for SLE patients (P less than .01). Coagulation protein C deficiency was found in 3 patients (17%). Even though within normal range, fibrinogen levels were significantly higher in SLE than in normal controls (219 versus 192 mg/dl, P less than .01) and plasminogen levels were significantly higher in SLE than in controls (117 versus 78.2%, P less than .01). Cross-linked fibrin derivatives (D-D dimers) were negative in all patients with SLE. Sixty-eight percent of SLE patients had high levels of antiphospholipid antibodies, but no correlation with the disturbances of the TPA/PAI system was found. It is concluded that most patients with SLE display severe abnormalities in the TPA/PAI anti-thrombotic system and that these abnormalities may be related to the lupus thrombophilia, apparently multifactorial in its origin.

Fibrinolysis↗
PubMed1993

[Pregnancy and systemic lupus erythematosus].

BACKGROUND: The interrelations between systemic lupus erythematosus (SLE) and pregnancy are reason for debate. Patients with SLE may present antiphospholipid antibodies (APL), anti-Ro (a-Ro) or anti-La (a-La) with their influence in pregnancy not being well defined. METHODS: The pregnancies in a series of 96 fertile age women with SLE, followed during the period of 1975-1991 with a mean follow up of 6.2 years were studied. The spontaneous or provoked abortions, prematurity and the number of flares of the disease during pregnancy and during the year after birth were analyzed. The number of flares was compared with that presented by a control group of women with SLE of similar clinical features followed over the same time period. RESULTS: Twenty-seven patients had 35 pregnancies with 23 full term pregnancies, 5 spontaneous abortions and 7 provoked abortions. Twenty-six percent of the deliveries were premature. One or two flares related with 43% of the pregnancies with viable fetus (11 flares in total) were registered. The number of flares in the control group was 6%. The difference was not statistically significant (p = 0.55) but the relative risk was of 1.6. No relation was found between the presence of APL, a-Ro and a-La and the course of the pregnancy. No case of lupus neonatorum was observed. CONCLUSIONS: Pregnancy is not advised in patients with systemic lupus erythematosus in remission. Patients should be more frequently controlled during pregnancy and post partum. The presence of antiphospholipid antibodies or anti-Ro does not modifies the prognosis.

Abortion, Induced↗
PubMed2006

Transforming growth factor beta 1 in children with systemic lupus erythematosus: a possible relation with clinical presentation of lupus nephritis.

Plasma and urinary (latent and active) TGF-beta1 levels were assessed in 32 children with active lupus and compared to 15 healthy controls of matched age and sex. Plasma latent and active TGF-beta1 levels in children with active disease were significantly lower than controls (P = 0.004 and P < 0.001 respectively). Plasma active TGF-beta1 correlated negatively with Systemic Lupus Erythematosus Disease Activity Index (r = -0.38, P = 0.03). On the contrary, urinary latent and active TGF-beta1 levels in children with active disease were significantly higher than controls (P < 0.001 and P = 0.003 respectively). Urinary active TGF-beta1 levels correlated positively with Anti-ds DNA titre (r = 0.42, P = 0.015) and negatively with serum C3 levels (r = -0.48, P = 0.005). Patients with symptomatic nephritis had significantly elevated urinary active TGF-beta1 levels in comparison to those with silent nephritis (P = 0.008). From this data we conclude that lowered plasma TGF-beta1 levels may be a feature of systemic immune dysfunction in children with active lupus while increased renal production of active TGF-beta1 seems to have a role in the clinical presentation of lupus nephritis.

Adolescent↗
PubMed2005

Subacute cutaneous lupus erythematosus: a paraneoplastic dermatosis?

Subacute cutaneous lupus erythematosus (SCLE) is characterized by clinical, laboratory and immunological features different from those of systemic lupus erythematosus (SLE). We describe the case of a patient with a 2-year history of SCLE that demonstrated a close temporal relationship with a squamous cell malignancy of the head and neck. This association has not been previously reported. We also review the evidence for SCLE as a 'paraneoplastic dermatosis' and discuss the criteria for diagnosis and possible pathogenesis.

Carcinoma, Squamous Cell↗
PubMed1984

Silent lupus nephritis among patients with discoid lupus erythematosus.

A kidney biopsy was performed in 7 hypocomplementemic discoid lupus erythematosus patients despite the absence of overt renal involvement. Five patients had glomerular immune deposits and 2 patients with disseminated discoid lupus erythematosus exhibited definite proliferative glomerulonephritis. Those findings show that silent lupus nephritis may be encountered in discoid as well as in systemic lupus erythematosus, providing additional evidence supporting the unity of the disease. We suggest that hypocomplementemic patients with discoid lupus erythematosus must be carefully screened for renal disease by periodic urinalysis examinations.

Complement C3↗
PubMed1992

Severe sun sensitivity and the presence of antinuclear antibodies in patients with polymorphous light eruption-like lesions. A form fruste of photosensitive lupus erythematosus?

BACKGROUND: Initial lesions seen in some cases of lupus erythematosus may simulate the clinical features of polymorphous light eruption. OBJECTIVE: To evaluate the significance of antinuclear antibody screening in patients with polymorphous light eruption or to find better methods to define patients at risk to have lupus erythematosus, we analyzed the data of 198 patients with polymorphous light eruption. METHODS: History, type, and persistence of skin lesions were reviewed, and serologic variables, the minimal erythema dose, and the ultraviolet action spectrum for inducing skin lesions were determined. RESULTS: The morphologic features of skin lesions and results of phototesting were consistent with previously published characteristics of patients with polymorphous light eruption. Twenty-eight (14%) had high titers of antinuclear antibody (greater than or equal to 1:80). We found a highly significant correlation between severe sun sensitivity and the presence of high antinuclear antibody titers. Of these patients with severe sun sensitivity and high titers of antinuclear antibody, in three the diagnosis of systemic lupus erythematosus could be established according to American Rheumatism Association criteria. CONCLUSION: These results suggest that a combination of severe sun sensitivity and high titers of antinuclear antibody may characterize a subset of sun-sensitive patients with some features of lupus erythematosus.

Adult↗
PubMed1978

Myocardial infarct in a child with systemic lupus erythematosus.

An unusual case of systemic lupus erythematosus (SLE) in a young child is reported with sudden death from myocardial infarction. The diagnosis of lupus erythematosus in this patient was made by renal biopsy at the age of 3 years. Atherosclerosis of the coronary arteries and aorta was found at autopsy with occlusion of the anterior descending branch of the left coronary artery. It is suggested that the vascular changes in this case were related to hypertriglyceridemia and prolonged prednisone therapy superimposed on a hypersensitivity vasculitis related to SLE.

Child, Preschool↗
PubMed1984

Renal cortical necrosis in systemic lupus erythematosus.

A patient with active systemic lupus erythematosus had acute and irreversible renal failure after an uncomplicated cholecystectomy, and renal biopsy established the diagnosis of acute cortical necrosis. Patients with lupus have a diathesis for intravascular coagulation, and the interactive effects of immune complex or endotoxin-induced vascular injury, change in platelet dynamics, impairment of fibrinolysis, alterations in the mononuclear phagocytic system, and/or concomitant use of corticosteroids may have singularly or in combination been important in generating this rare renal complication of systemic lupus erythematosus in our patient.

Acute Disease↗