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PubMed1999

[A case report; systemic lupus erythematosus associated with lupus laryngitis].

Systemic lupus erythematosus (SLE) is an autoimmune collagen vascular disease which produces widespread damage to multiple organs. Few studies on laryngeal involvement in SLE have been reported. We report here a case of SLE complicated by lupus laryngitis. A 27-year-old woman was diagnosed as having SLE in October 1996 based on findings of polyarthritis, lymphocytopenia, positive anti-nuclear antibody and anti-Sm antibodies. Polyarthralgia disappeared transiently, and no other clinical symptoms were found. Therefore, she was followed with no medication. She presented hoarseness with high fever and facial erythema in September 1998. Laboratory findings on admission revealed pancytopenia and hypocomplementemia. Anti-nuclear antibody, anti-Sm antibody and anti-RNP antibody were all positive with high titers. Chest X ray examination showed pleural effusion. Laryngoscopy showed a bamboo-joint-like lesion at the middle of the bilateral vocal cords. She was treated with 60 mg of prednisolone (i.v.) which resulted in improvement of hoarseness, pancytopenia, hypocomplementemia and pleuritis. Based on these findings, we diagnosed her hoarseness as the manifestation of laryngitis associated with SLE (lupus laryngitis).

Adult↗
PubMed2004

Soluble adhesion molecule levels, neuropsychiatric lupus and lupus-related damage.

Nervous system dysfunction may occur in as many as 80% of patients with Systemic Lupus Erythematous (SLE) at some point in their disease course. Upregulation of adhesion molecules has been linked to acute SLE-related disease activity and chronic damage. We evaluated the relationship between soluble adhesion molecule levels and neuropsychiatric lupus (NPSLE) manifestations using the American College of Rheumatology (ACR) case definitions to investigate for evidence of a link between upregulation of adhesion molecules and NPSLE manifestations. Sera from the initial study visit of 133 SLE patients enrolled in the San Antonio Lupus Study of Neuropsychiatric Disease (SALUD) and 40 controls were evaluated for soluble adhesion molecule levels (VCAM-1, ICAM-1 and E-selectin) and antiphospholipid antibodies. A subset of 57 SLE patients were evaluated for soluble adhesion molecule levels and antiphospholipid antibodies on two subsequent study visits, as well. NPSLE manifestations at the time of sera ascertainment were recorded using ACR case definitions and SLE-related acute activity and damage were measured. Elevated levels of all three soluble adhesion molecules were seen in SLE patients compared to normal control values. Soluble VCAM-1 levels correlated with measures of current disease activity, NPSLE manifestations and deep venous thrombosis. Persistently positive levels of ICAM-1 and VCAM-1, but not E-selectin were association with increased SLE-related damage. Elevated levels of all soluble adhesion molecule levels correlated with abnormal levels of antiphospholipid antibodies, which are associated with some NPSLE manifestations and have been shown to upregulate adhesion molecule expression.

Adult↗
PubMed1996

Thrombosis and systemic lupus erythematosus: the Hopkins Lupus Cohort perspective.

Vascular damage in systemic lupus erythematosus (SLE) occurs through vasculitis, premature atherosclerosis, and hypercoagulability (predominantly due to the antiphospholipid antibody syndrome). In the Hopkins Lupus Cohort, a prospective cohort study, the incidence of thrombosis is 2 per 100 person-years of follow-up. Markers of immune-complex mediated injury (high anti-dsDNA and low C3), atherosclerosis (hypertension, hyperlipidemia, homocysteine) and antiphospholipid antibodies (lupus anticoagulant or anticardiolipin) are independent predictors of thrombosis. Hydroxychloroquine use is protective against future thrombosis.

Cohort Studies↗
PubMed1983

Lupus cystitis: primary bladder manifestations of systemic lupus erythematosus.

Clinical and radiographic findings of cystitis are reported in six patients with systemic lupus erythematosus. All patients had clinical manifestations of cystitis; radiography showed the bladder capacities were reduced and the bladder walls were thickened and irregular. Ureterohydronephrosis was found in some patients. Abnormal clinical and roentgenographic findings were reversed with high-dose corticosteroid therapy. These patients had concomitant widespread multisystem manifestations, especially of the central nervous system and gastrointestinal tract. No explanation for the bladder findings could be found other than lupus erythematosus. Urinary bladder involvement may be a primary manifestation of systemic lupus erythematosus, sometimes overshadowed by other major organ system disease. Corticosteroids appear to reverse most of the acute manifestations but the long-term prognosis is not known and may not be favorable.

Adrenal Cortex Hormones↗
PubMed2004

Lupus tumidus: a variant of cutaneous lupus erythematosus.

Lupus tumidus is a rare sub-type of chronic cutaneous lupus erythematosus characterized by dermal plaques in which excessive mucin accumulates early in disease process. We report a middle aged women having succulent, edematous and persistent plaque over her face for five years that was not responding to various empirical treatments offered to her. Finally, on clinico-pathological basis, it was diagnosed as a case of tumid lupus erythematosus (TLE) and she responded satisfactorily to the treatment regimen including oral steroids, chloroquine and application of sun screen.

Adult↗
PubMedDate not supplied

Lupus erythematosus tumidus and chronic discoid lupus erythematosus in carriers of X-linked chronic granulomatous disease.

Two Caucasian carriers for chronic granulomatous disease (CGD) developed cutaneous lupus erythematosus (LE) with clinically and morphologically characteristic appearance for chronic discoid lupus erythematosus (DLE) and lupus erythematosus tumidus (LET). Direct immunofluorescent examinations and ANA titers were positive in both young women. No systemic involvement due to the ACR criteria was evident. Their sons suffered from X-linked cytochrome-b negative CGD. The diagnosis of CGD was based on measurement of oxidative burst activity by nitroblue tetrazolium (NBT) slide test and by flow cytometry using dihydrorhodamine 123 (DHR). The absence of cytochrome b558 in neutrophilic granulocytes was confirmed photometrically and by flow cytometry using the 7D5 monoclonal antibody against cytochrome b. We report for the first time the association of the photosensitive LE subtype LET and the X-linked CGD carrier state. Tissue damage by UV radiation and a reduced antimicrobial capacity may lead to recurrent immune stimulation and may together with genetic predisposition explain the occurrence of cutaneous LE in female carriers of CGD.

Adult↗
PubMed2001

Quantitative analysis of interstitial mast cells in lupus and non-lupus membranous glomerulopathy.

Eleven renal biopsy specimens from patients with lupus membranous glomerulopathy (LMGN) and 16 from patients with primary (nonlupus) membranous glomerulopathy (NLMGN) for whom light, electron microscopy and immunofluorescence microscopy, and full clinical data were available were examined quantitatively. As a control 10 biopsy specimens of the kidneys removed because of trauma were used. Morphometric investigations were performed by means of a computer image analysis system to evaluate whether mast cells have a role in tubulointerstitial fibrosis in lupus and nonlupus membranous glomerulopathy and to examine the relationship between mast cells and interstitial alpha-smooth muscle actin (alpha-SMA) expression as well as interstitial infiltrates. The morphometric study revealed that the mean values of interstitial tryptase positive cells, expression of alpha-SMA, interstitial volume, CD68+, CD45RB+, CD43+ and CD20+ cells were significantly increased in LMGN as compared with NLMGN. In both LMGN and NLMGN groups there were significant positive correlations between interstitial tryptase positive cells and interstitial expression of alpha-SMA, interstitial volume, serum creatinine as well as CD68+ cells. The present data suggest that in cases of membranous glomerulopathy with a large number of interstitial mast cells systemic lupus erythematosus should be taken into consideration, even if this aetiology was not clinically suggested at the time of biopsy. Additionally, in both LMGN and NLMGN significant positive correlations between interstitial mast cell count and relative interstitial volume support the role of these cells in the development of interstitial fibrosis, however this relationship needs further investigations.

Adult↗
PubMed2002

Organ manifestations influence differently the responsiveness of 2 lupus disease activity measures, according to patients' or physicians' evaluations of recent lupus activity.

OBJECTIVE: To determine (1) which organ system manifestations contribute to the overall responsiveness of the Systemic Lupus Activity Measure (SLAM, revised 1991 with minor modifications as SLAM-R) and the Systemic Lupus Erythematosus Disease Activity Index (SLEDAI); and (2) whether responsive items differ for physicians and patients. METHODS: Blinded data were obtained from repeated visits of 76 patients in the Study of Methotrexate in Lupus Erythematosus. At each visit, physicians and patients reported improvement, no change, or deterioration, and physicians then completed SLAM-R and SLEDAI. Items in SLAM-R and SLEDAI were grouped by organ system. The generalized estimating equations approach was used to measure associations between change in organ system activity and physician or patient perception of change in overall disease activity. The outcomes assessed, in separate analyses, were improvement and deterioration from the previous visit. RESULTS: Seventy-six patients contributed a total of 471 observations. The strongest correlates of physician-reported improvement were decreased constitutional, gastrointestinal (GI), and musculoskeletal involvement (components of SLAM-R), and decreased musculoskeletal (MSK) and central nervous system involvement (SLEDAI). Improvement reported by patients was most strongly associated with decreases in erythrocyte sedimentation rate and MSK and reticuloendothelial activity (SLAM-R), and in MSK activity (SLEDAI). Increased integument and MSK subscores (SLAM-R) and serosal and MSK subscores (SLEDAI) were associated with overall deterioration reported by physicians. Patient-reported deterioration was associated with increased GI subscores (SLAM-R) and with no changes in organ system involvement in SLEDAI. CONCLUSION: Organ systems associated with reported change in overall SLE activity differed between SLAM-R and SLEDAI, between patients and physicians, and between each direction of change.

Adult↗
PubMed2002

Defining lupus cases for clinical studies: the Boston weighted criteria for the classification of systemic lupus erythematosus.

OBJECTIVE: The 1982 American College of Rheumatology (ACR) revised criteria for the classification of systemic lupus erythematosus (SLE), updated in 1997, have become the standard for establishing eligibility of subjects for epidemiologic and clinical lupus studies. These criteria may exclude patients with limited disease, restricting the generalizability of research findings. We developed and evaluated the ability of a weighted classification system to identify a broader spectrum of patients with lupus. METHODS: We constructed the Boston Weighted Criteria system for the classification of SLE, updating that developed in 1984. Using a hospital billing database, we identified 27l patients seen in our rheumatology clinic for possible SLE and reviewed medical records for all ACR criteria and the treating rheumatologist's diagnosis. We compared both the Boston Criteria and the treating rheumatologist's diagnosis to the updated 1982 ACR criteria; we also compared the Boston Criteria to the treating rheumatologist's diagnosis. RESULTS: The Boston Criteria identified 190/271 patients as having SLE, the rheumatologist's diagnosis identified 179/271, and the ACR criteria identified 171/271. The Boston Criteria had a sensitivity of 93% and specificity of 69% compared to the ACR criteria, and would identify 7% more patients. CONCLUSION: The Boston Criteria identify a larger number of patients compared with the current ACR criteria, while retaining face validity. This reflects the inclusion of patients with objective findings of SLE but less than 4 ACR criteria. Our Boston Criteria system could minimize selection bias and increase the generalizability of clinical SLE studies.

Adolescent↗
PubMed2004

Transverse myelitis as the first manifestation of systemic lupus erythematosus or lupus-like disease: good functional outcome and relevance of antiphospholipid antibodies.

OBJECTIVE: Transverse myelitis (TM) is a rare complication of systemic lupus erythematosus (SLE). Although usually a late manifestation of SLE, it can occur at presentation. We investigated the clinical presentation, treatment and outcome of 15 patients with TM as the presenting manifestation of SLE or lupus-like disease. METHODS: All patients received corticosteroids, while 13 also received immunosuppressive therapy. Five patients were fully anticoagulated with warfarin. RESULTS: A sensory level with spastic lower limb weakness and sphincter disturbance was the most common presentation: 14/15 patients had a thoracic or cervical sensory level. Cerebrospinal fluid examination showed high protein concentrations in 3 patients and oligoclonal bands in 8. Eleven of the 15 (73%) had antiphospholipid antibodies (aPL). Of the 15 patients, 3 had complete resolution of the symptoms, 6 had good functional improvements, 5 had good to fair outcome with some functional deficit, and one patient who received corticosteroids alone later died from pneumonia. CONCLUSION: We describe 15 patients with TM as the presenting manifestation of SLE or lupus-like disease with a high prevalence of aPL. Our data support the view that early diagnosis and immunosuppressive therapy may be superior to corticosteroids alone in improving functional outcome. In those patients with aPL, antiplatelet agents and/or warfarin should also be considered.

Adult↗
PubMed1989

Budd Chiari syndrome, visceral arterial occlusions, recurrent fetal loss and the "lupus anticoagulant" in systemic lupus erythematosus.

A 30-year-old Chinese woman had been diagnosed as having systemic lupus erythematosus (SLE) at age 25 after 4 spontaneous abortions, all between 8 and 12 weeks of gestation. A year later she developed mild jaundice and at laparotomy, nodular enlargement of the right lobe of the liver and splenomegaly were seen. She was found to have a "lupus anticoagulant" and false positive test for syphilis (VDRL), but persistently negative antibodies to cardiolipin. Angiography demonstrated occlusion and stenoses of visceral abdominal arteries and hepatic venography also showed evidence of hepatic venous occlusions. She subsequently developed thyrotoxicosis which was treated with carbimazole, followed by radioactive iodine. The SLE and lupus anticoagulant were treated with systemic steroids (prednisolone) and anticoagulation (warfarin).

Abortion, Spontaneous↗
PubMed1987

Activities of dipeptidyl peptidase II and dipeptidyl peptidase IV in mice with lupus erythematosus-like syndrome and in patients with lupus erythematosus and rheumatoid arthritis.

We examined the activities of peptidases in plasma and tissues of the New Zealand Black (NZB) mouse as an animal model of human systemic lupus erythematosus, and also in serum from patients with rheumatoid arthritis and systemic lupus erythematosus. Activities of dipeptidyl peptidase II (DAP II) and post-proline cleaving enzyme (PPCE) were increased, and dipeptidyl peptidase IV (DAP IV) activity was decreased in plasma and spleen of NZB mice, as compared with the control BALB/c mice. Likewise, the activity of DAP II was increased and that of DAP IV was decreased in serum of patients with rheumatoid arthritis and systemic lupus erythematosus. These results indicate the importance of hydrolytic enzymes in the pathogenesis of autoimmune diseases.

Animals↗
PubMed1987

[Lupus associated membrane protein. Changes in the sensitivity of proteases during the life span of mice with lupus].

A murine monoclonal anti-DNA antibody, PME77, spontaneously produced in autoimmune B/W mouse, has been shown to react with a protein present at the surface of several cells involved in lupus pathogenesis. We have called this cell-surface protein LAMP (Lupus Associated Membrane Protein). Mild elastase treatment of lymphoid cells from non autoimmune (BALB/c or CBA/ca) mice releases five polypeptides (34, 33, 17, 16 and 14 kDa) recognized by PME77. These polypeptides are not found after treatment of these cells with papain or trypsin. When lymphoid cells from autoimmune mice (MRL/lpr/lpr and B/W) are treated with elastase, trypsin or papain, PME77 detected in all supernatants a single polypeptide of 55 kDa. It is demonstrated in the present work that: (1) this 55 kDa polypeptide is also detected in the elastase supernatant of glomeruli from MRL/lpr/lpr and B/W mice but not from BALB/c and CBA/ca mice. These results suggest that LAMP expressed at the surface of lymphoid and glomerular cells from lupus mice displays altered sensitivity to proteases. (2) The change in sensitivity to proteolytic enzymes appears between 1 and 3 weeks after birth in MRL/lpr/lpr mice. Such modifications might results in the appearance of a non-self antigen and elicit an anti-LAMP immune response.

Animals↗
PubMed1976

[Diagnostic problems associated with the coincidence of bullous dermatosis and lupus erythematosus visceralis. Demonstration of a case with Duhring's dermatitis herpetiformis in association with visceral lupus erythematosus].

This report describes a patient, whom we observed for 3 years, exhibiting a coincidence of dermatitis herpetiformis Duhring and systemic Lupus erythematosus. The difficulties involved in demarcating bullous lupus erythematosus from the more rare association of bullous pemphigoid or dermatitis herpetiformis with systemic lupus erythematosus are discussed in connection with differential, histological, direct and indirect immunofluorescence diagnostic aids.

Adult↗
PubMed1975

[Morphologic changes in the chorio-allantoic membrane and organs of chick embryos inoculated with blood of patients with systemic lupus erythematosus (concerning the viral etiology of systemic lupus erythematosus)].

Morphological studies of 14 chorionallantoic membranes and organs of chick embryos which had been infected on the 7--8th day of incubation with leucocytic mass from patients with systemic lupus erythematosus were carried out. Corresponding virological and morphological control studies were performed. The changes observed were repeatedly reproduced in multiple inoculations (from the 2nd to the 16th passage). Apart from morphological changes, which may be listed among general pathological processes (impairment of the circulation, dystrophy, proliferation), changes testifying to a possible cytopathic effect disintegration of nuclei, formation of gigantic cells and multinuclear symplasts were also noted. The most pronounced changes, both in the chorionallantoic membrane and in internal organs, were observed on the 3rd--5th passage and on the 3rd--5th day of incubation following inoculation. They correlated with the clinical activity of the process in patients from whom the blood for inoculation had been taken. The data obtained justify the assumption concerning the existence in the blood of the patients with systemic lupus erythematosus a transplantable agent producing a cytopathic action, and may be considered as a new indirect corroborative evidence in favour of the concept of the viral nature of systemic lupus erythematosus.

Animals↗
PubMedDate not supplied

Systemic lupus erythematosus relapse with lupus cystitis.

A 45-year-old woman who had been diagnosed as having systemic lupus erythematosus (SLE) at the age of 28 years and who had been in remission developed severe urinary frequency, watery diarrhea, vomiting and weight loss. She also developed acute renal failure and her serological examination was consistent with active SLE. She had a markedly decreased urinary bladder capacity of 20 ml with hydroureteronephrosis. Histopathological study of her urinary bladder biopsy specimen showed mucosal edema, infiltration by lymphocytes and granulocytes, and deposition of IgA in the epithelium and submucosal region. We diagnosed this as a case of lupus cystitis. The patient's symptoms were alleviated by bilateral nephrostomy and corticosteroid therapy. In the present episode the patient showed none of the usual symptoms of SLE. This case and others reported in the literature show that lupus cystitis presents with specific signs and symptoms and therefore, this syndrome may represent a specific clinical manifestation of SLE.

Acute Kidney Injury↗