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A Case of Subacute Cutaneous Lupus Erythematosus in a Patient with Mixed Connective Tissue Disease: Successful Treatment with Plasmapheresis and Rituximab  [PDF]
M. Fantò,S. Salemi,F. Socciarelli,A. Bartolazzi,G. A. Natale,I. Casorelli,A. Pavan,S. Vaglio,R. Di Rosa,R. D'Amelio
Case Reports in Rheumatology , 2013, DOI: 10.1155/2013/857694
Abstract: A 30-year-old woman affected by Mixed Connective Tissue Disease with scleroderma spectrum developed a facial eruption, a clinical and histological characteristic of subacute cutaneous lupus erythematosus (SCLE). Speckled anti-nuclear antibodies, high-titer anti-ribonucleoprotein1, anti-Sm, anti-Cardiolipin (aCL) IgG/IgM, and anti-Ro/SSA antibodies were positive. SCLE was resistant to Azathioprine, Hydroxychloroquine, and Methotrexate while Mycophenolate Mofetil was suspended due to side effects. Subsequently, the patient was treated with three cycles of therapeutic plasma exchange (TPE) followed, one month after the last TPE, by the anti-CD20 antibody Rituximab (RTX) (375?mg/m2 weekly for 4 weeks). Eight and 16 months later the patient received other two TPE and RTX cycles, respectively. This therapeutic approach has allowed to obtain a complete skin healing persistent even after 8-month follow-up. Moreover, mitigation of Raynaud's phenomenon, resolution of alopecia, and a decline of aCL IgG/IgM and anti-Ro/SSA antibodies were observed. 1. Introduction Mixed Connective Tissue Disease (MCTD) is currently defined as an overlapping syndrome with clinical features of Systemic Sclerosis (SSc), Systemic Lupus Erythematosus (SLE), Rheumatoid arthritis (RA), and Polymyositis/Dermatomyositis (PM/DM); the presence of high-titer anti-ribonucleoprotein1 (U1RNP) or speckled anti-nuclear antibodies (ANA) at titer ≥1?:?2,000 is necessary for the diagnosis. The disease affects mainly women in the third decade of life (from 80 to 90%) but it has been also reported in children and in over-80-year-old people [1]. The most frequent clinical manifestations are Raynaud's phenomenon (RP), swollen hands, sclerodactyly, arthritis, myalgias, and oesophageal dysmotility, and also alopecia, malar rash, lymphadenopathy, or kidney damage can be present. Rarely, subacute cutaneous lupus erythematosus (SCLE), characterized by annular or papulosquamous lesions, photosensitivity, and presence of anti-Ro/SSA and anti-La/SSB antibodies, has been described in MCTD patients [2, 3]. MCTD therapy should be identified for each patient depending on the affected organ, but generally there is a good response to steroids, different types of vasodilators, and immunosuppressive agents such as Hydroxychloroquine (HCQ), Azathioprine (AZA), Methotrexate (MTX), or Cyclophosphamide (CYC) [1]. 2. Case Presentation A case of a thirty-year-old woman affected by MCTD with scleroderma spectrum and epilepsy since she was fifteen is here reported. At the beginning she presented fever up to 40°C, arthalgias
Apoptosis and Redistribution of the Ro Autoantigen in Balb/c Mouse Like in Subacute Cutaneous Lupus Erythematosus  [PDF]
Rafael Herrera-Esparza,Ricardo Villalobos,Juan-Jose Bollain-y-Goytia,Roxana Ramírez-Sandoval,Sergio H. Sánchez-Rodriguez,Guadalupe Pacheco-Tovar,Esperanza Avalos-Diaz
Clinical and Developmental Immunology , 2006, DOI: 10.1080/17402520600876796
Abstract: In subacute cutaneous lupus eryhematosus (SCLE) the cutaneous antigens constitute the main source of Ro and La autoantigens. The aim of this investigation was to demonstrate if UV light increases the availability of Ro autoantigen in the skin, also the blocking effect of Ac-DEVD-CMK a caspase inhibitor was assessed. For this purpose newborn Balb/c mice were UVB irradiated (5–30 mJ/cm2) equivalent to a moderate to severe sunburn. Animals were injected with monoclonal anti-Ro antibodies from SCLE patients. Apoptosis was also induced by anti-Fas antibody injection. Skin samples were examined by direct immunofluoresence, by TUNEL, and the expression of caspase 3 by RT-PCR. Major findings of present studies were: 1. UVB irradiation and anti-Fas induced apoptosis of keratinocytes. 2. Apoptosis redistribute the Ro antigen on cell surface and is better triggered by Ro antibody. 3. The caspase 3 inhibitor Ac-DEVD-CMK decreases the availability of Ro autoantigen in epidermis and prevents deposition of anti-Ro. In conclusion, the caspase pathway would be blocked to avoid anti-Ro deposition along skin; this finding would be a prospect in the treatment of SCLE patients.
Diffuse Leukoencephalopathy and Subacute Parkinsonism as an Early Manifestation of Systemic Lupus Erythematosus  [PDF]
Gary G. Tse,Alberto S. Santos-Ocampo,Dominic C. Chow,Aaron M. McMurtray,Beau K. Nakamoto
Case Reports in Neurological Medicine , 2013, DOI: 10.1155/2013/367185
Abstract: Parkinsonism in SLE is rare. Diffuse leukoencephalopathy is equally uncommon and is associated with a poor prognosis. We present a single case of a 50-year-old Filipino man who presented with a generalized discoid rash after starting lisinopril. The rash persisted despite discontinuation of lisinopril, and over the next three months, he developed rapidly progressive parkinsonism. Brain MRI showed symmetric confluent T2-hyperintensities involving the white matter and basal ganglia. Four of the 11 American College of Rheumatology criteria for the classification of SLE were met. A rheumatologist made a diagnosis of SLE with cutaneous and central nervous system involvement. Significant neurologic and radiologic improvement occurred following treatment with IV steroids followed by a prolonged taper. This report highlights a case of subacute parkinsonism with a diffuse leukoencephalopathy as an early manifestation of SLE which resulted in a good recovery following treatment with only immunosuppressive therapy. 1. Introduction Central nervous system (CNS) involvement is present in up to 70% of individuals with systemic lupus erythematosus (SLE) and can present with an acute confusional state, demyelinating syndrome, stroke, seizures, or cognitive dysfunction [1]. Radiologic abnormalities associated with CNS lupus include demyelinating plaques, myelitis, ischemic or hemorrhagic stroke, dural venous sinus thrombosis, rhombencephalitis, and cerebral atrophy [2]. We report a single case of subacute parkinsonism with a diffuse leukoencephalopathy on brain MRI as an early manifestation of SLE. 2. Report of a Case A 50-year-old Filipino male with a history of hypertension developed a discoid, photosensitive, and hyperpigmented rash on sun-exposed areas of his face, arms, and legs in March 2012 shortly after starting lisinopril. Lisinopril was discontinued in April 2012 without resolution of the rash, prompting consultation with a dermatologist who suspected discoid lupus and ordered screening lupus labs. Complete blood count (CBC) was normal. Erythrocyte sedimentation rate (ESR) was 76?mm/hour, anti-nuclear antibody (ANA) 1?:?320 in a homogenous and fine speckled pattern, anti-histone and anti-RNP antibodies were positive, and complement C50, C3, and C4 were low. Anti-double-stranded DNA (anti-ds DNA) and anti-smith (anti-Sm) antibodies were negative. Initial impression was drug-induced lupus. In July 2012, he was admitted for recurrent falls. Neurological examination was notable for reduced degree of facial expression (hypomimia), weak and soft speech (hypophonia),
Subacute cutaneous lupus erythematosus
Singh R,Singh Sanjay,Pandey S
Indian Journal of Dermatology, Venereology and Leprology , 1995,
Cutaneous Manifestations of Systemic Lupus Erythematosus  [PDF]
Luís Uva,Diana Miguel,Catarina Pinheiro,Jo?o Pedro Freitas,Manuel Marques Gomes,Paulo Filipe
Autoimmune Diseases , 2012, DOI: 10.1155/2012/834291
Abstract: Systemic lupus erythematosus (SLE) is a multiorgan autoimmune disease of unknown etiology with many clinical manifestations. The skin is one of the target organs most variably affected by the disease. The American College of Rheumatology (ACR) established 11 criteria as a classificatory instrument to operationalise the definition of SLE in clinical trials. They were not intended to be used to diagnose individuals and do not do well in that capacity. Cutaneous lesions account for four of these 11 revised criteria of SLE. Skin lesions in patients with lupus may be specific or nonspecific. This paper covers the SLE-specific cutaneous changes: malar rash, discoid rash, photosensitivity, and oral mucosal lesions as well as SLE nonspecific skin manifestations, their pathophysiology, and management. A deeper thorough understanding of the cutaneous manifestations of SLE is essential for diagnosis, prognosis, and efficient management. Thus, dermatologists should cooperate with other specialties to provide optimal care of SLE patient. 1. Introduction The nosographic concept of lupus erythematosus (LE) includes 3 major subtypes: chronic cutaneous LE, subacute cutaneous LE, and systemic or acute cutaneous LE. Besides these 3 subtypes, other less frequent clinical varieties may occur [1]. Systemic lupus erythematosus (SLE) is a multiorgan autoimmune disease of unknown etiology that can have many clinical manifestations (Table 1). The skin is involved in up to 85% of systemic lupus erythematosus (SLE) cases and may be the only organ involved in cutaneous lupus erythematosus (CLE). Table 1: Cutaneous manifestations of SLE. The diagnosis of the cutaneous manifestations of LE is based on clinical, histopathology, and immunohistology of skin lesions. In addition, serum autoantibodies are considered immunologic markers for distinct clinical types of the illness. The Cutaneous Lupus Erythematosus Disease Area and Severity Index (CLASI) is used as a clinical tool that standardizes the way disease activity is described and provides guidelines for identifying a clinical change. This clinical tool quantifies disease activity and damage in cutaneous lupus erythematosus. The activity score is based on the erythema, scale, mucous membrane lesions, and nonscarring alopecia. A recent study gives us a foundation for the practical use of the CLASI in clinical trials as a tool to measure disease severity and responsiveness to therapy [2]. In 1982, the diagnosis criteria for SLE were published by the American College of Rheumatology (ACR) which were revised in 1997 and are currently
Eyelid involvement in disseminated chronic cutaneous lupus erythematosus  [cached]
Pandhi D,Singal A,Rohtagi J
Indian Journal of Dermatology, Venereology and Leprology , 2006,
Abstract: Eye involvement in chronic cutaneous lupus erythematosus (CCLE) has been reported infrequently. We describe two cases of disseminated CCLE, a 50-year-old woman and a 45-year-old man, with eyelid involvement and disease duration of 4 and 20 years respectively. The first patient showed an excellent response to chloroquine while there was lack of response in the second patient.
Cutaneous neonatal lupus erythematosus with unusual features  [cached]
Sawant Shankar,Amladi S,Wadhawa S,Nayak C
Indian Journal of Dermatology, Venereology and Leprology , 2007,
Abstract: A three month-old boy was brought by his mother with complaints of multiple reddish lesions on his trunk and face since birth. The patient had erythematous annular plaques with scaling on his extremities, palms and soles with periorbital erythema and edema giving the characteristic "eye mask" or "owl′s eye" appearance. His mother did not have history of any illness. Hemogram, liver and renal function tests were within normal limits. A skin biopsy was suggestive of subacute cutaneous lupus erythematosus. Immunological work-up was positive for antinuclear antibodies (ANA) (1:40) with anti-Ro titers of 3.4 and 3.47 (>1.1 = clinically significant titre) in the mother and child respectively, although negative for anti-La antibodies. The child′s electrocardiogram and 2D echocardiography were normal. We are presenting a case of anti-Ro-positive cutaneous lupus erythematosus with an uncommon skin manifestation.
Verrucous cutaneous lupus erythematosus  [cached]
Thappa Devinder,Venkateswaran Sri,Reddy B,Garg B
Indian Journal of Dermatology, Venereology and Leprology , 1995,
Abstract: An unusual case of cutaneous lupus erythematous with verrucous lesions is reported for its rarity and clinical interest.
Cutaneous manifestations of systemic lupus erythematosus in a tertiary referral center
Kole Alakes,Ghosh Alakendu
Indian Journal of Dermatology , 2009,
Abstract: Background : Systemic lupus erythematosus (SLE) is an autoimmune disease with multiorgan involvement. The skin is the second most commonly affected organ. SLE with skin lesions can produce considerable morbidity resulting from painful skin lesions, alopecia, disfigurement, etc. Skin lesions in patients with lupus may be specific (LE specific) or may be non specific (LE non specific). Acute cutaneous LE (Lupus specific) has a strong association with systemic disease and non-specific skin lesions always indicate disease activity for which patients present to rheumatologists and internists. Therefore, a thorough understanding of the cutaneous manifestations of SLE is essential for most efficient management. Aims: The aims of this study were to evaluate the patterns and prevalence of skin lesions in patients with SLE and to assess the relationship between skin lesions and other systemic involvement. Materials and Methods: At the Department of Rheumatology and Clinical Immunology, IPGME&R in Kolkata, 150 patients with SLE fulfilling the clinical and laboratory criteria of the American Rheumatology Association (updated 1982) were examined and followed-up for cutaneous manifestations between January 2002 and January 2007. Results: Skin lesions were important clinical features. About 45 patients (30%) presented with skin lesions although all patients had skin lesions during the follow-up period. Skin changes noted were as follows: Lupus specific lesions: malar rash in 120 patients (80%), photosensitive dermatitis in 75 patients (50%), generalized maculopapular rash in 40 patients (26.67%), discoid rash in 30 patients (20%), subacute cutaneous lupus erythematosus (SCLE) in 5 patients (3.34%), lupus profundus in 5 patients (3.34%). The lupus non-specific lesions were non-scarring alopecia in 130 patients (86.67%), oral ulcers in 85 patients (56.67%), vasculitic lesions in 50 patients (33.34%), bullous lesions in 15 patients (10%), Raynaud′s phenomenon in 10 patients (6.67%), pyoderma gangrenosum in 2 patients (1.34%), erythema multiforme in 10 patients (6.67%), and nail fold infarcts in 2 patients (1.34%); however, mucosal discoid lupus, lichenoid discoid lupus, livedo reticularis, sclerodactyly, etc. were not detected. Patients having lupus-specific skin lesions e.g., malar rash were associated with systemic involvement, whereas those having lupus non-specific skin lesions were associated with disease flare. Conclusions: Skin lesions in patients with SLE are important disease manifestations and proper understanding is essential for diagnosis and efficient manage
Polymorphisms of the ITGAM Gene Confer Higher Risk of Discoid Cutaneous Than of Systemic Lupus Erythematosus  [PDF]
Tiina M. J?rvinen,Anna Hellquist,Sari Koskenmies,Elisabet Einarsdottir,Jaana Panelius,Taina Hasan,Heikki Julkunen,Leonid Padyukov,Marika Kvarnstr?m,Marie Wahren-Herlenius,Filippa Nyberg,Mauro D'Amato,Juha Kere,Ulpu Saarialho-Kere
PLOS ONE , 2012, DOI: 10.1371/journal.pone.0014212
Abstract: Lupus erythematosus (LE) is a heterogeneous disease ranging from mainly skin-restricted manifestations (discoid LE [DLE] and subacute cutaneous LE) to a progressive multisystem disease (systemic LE [SLE]). Genetic association studies have recently identified several strong susceptibility genes for SLE, including integrin alpha M (ITGAM), also known as CD11b, whereas the genetic background of DLE is less clear.
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