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Pemphigus Foliaceus

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81. Elevated serum eosinophil cationic protein and transforming growth factor-α levels in a patient with pemphigus vegetans. (PubMed)

Elevated serum eosinophil cationic protein and transforming growth factor-α levels in a patient with pemphigus vegetans. Pemphigus vegetans (PVeg) is a rare variant of pemphigus, characterized by vegetating lesions mainly with antidesmoglein 3 antibodies. However, the pathomechanisms for PVeg is still unknown. We present a patient with PVeg mainly associated with antidesmocollin (Dsc)3 antibodies, who originally developed pemphigus foliaceus. Serum levels of eosinophil cationic protein

2018 Clinical & Experimental Dermatology

82. Coexistent Solid Malignancies in Pemphigus: A Population-Based Study. (PubMed)

Coexistent Solid Malignancies in Pemphigus: A Population-Based Study. The association of pemphigus vulgaris and pemphigus foliaceus with comorbid malignancies is yet to be firmly established.To estimate the association between pemphigus and a wide range of nonhematologic malignancies using one of the largest cohorts of patients with pemphigus to date.For this cross-sectional study, we used the computerized database of Clalit Health Services, the largest public health care provider organization (...) in Israel insuring 4.4 million individuals in the settings of general community clinics, primary care and referral centers, and ambulatory and hospitalized health care. The study included 1985 patients with pemphigus and 9874 control patients and was conducted from January 2004 to December 2014.The prevalence of 17 different solid malignancies was compared between patients diagnosed with pemphigus and age-, sex-, and ethnicity-matched control patients; χ2 and t tests were used for univariate analysis

2018 JAMA dermatology (Chicago, Ill.)

83. A prospective study comparing patients with early and late relapsing pemphigus treated with rituximab. (PubMed)

A prospective study comparing patients with early and late relapsing pemphigus treated with rituximab. Rituximab (RTX) is an effective therapy for patients with pemphigus; however, the therapy does not prevent relapse.To compare early relapsing patients (before 12 months) and late relapsing patients (after 24 months) following RTX therapy.In this prospective study, 19 patients were enrolled (14 with pemphigus vulgaris and 5 with pemphigus foliaceus). The baseline disease score, autoantibody (...) levels, and percentage of CD20+ cells of patients with pemphigus were measured. Patients received 1 cycle of RTX and were followed for 26 months.Among early relapsing patients (n = 5), the time to relapse was 6 to 11 months. Among late relapsing patients (n = 6), the time to relapse was 24 to 26 months. A significant difference was observed in the mean baseline anti-desmoglein 1 (DSG1) index between early relapsing (705.72) and late relapsing patients (210.4) (P = .0014). A significant negative

2018 Journal of American Academy of Dermatology

84. Keratins Regulate p38MAPK-Dependent Desmoglein Binding Properties in Pemphigus (PubMed)

Keratins Regulate p38MAPK-Dependent Desmoglein Binding Properties in Pemphigus Keratins are crucial for the anchorage of desmosomes. Severe alterations of keratin organization and detachment of filaments from the desmosomal plaque occur in the autoimmune dermatoses pemphigus vulgaris and pemphigus foliaceus (PF), which are mainly caused by autoantibodies against desmoglein (Dsg) 1 and 3. Keratin alterations are a structural hallmark in pemphigus pathogenesis and correlate with loss (...) of intercellular adhesion. However, the significance for autoantibody-induced loss of intercellular adhesion is largely unknown. In wild-type (wt) murine keratinocytes, pemphigus autoantibodies induced keratin filament retraction. Under the same conditions, we used murine keratinocytes lacking all keratin filaments (KtyII k.o.) as a model system to dissect the role of keratins in pemphigus. KtyII k.o. cells show compromised intercellular adhesion without antibody (Ab) treatment, which was not impaired further

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2018 Frontiers in immunology

85. The Usefulness of Indirect Immunofluorescence in Pemphigus and the Natural History of Patients With Initial False-Positive Results: A Retrospective Cohort Study (PubMed)

diagnosed with pemphigus vulgaris (PV) and 29 patients with pemphigus foliaceus (PF). The sensitivity of this immunoassay was significantly higher for the diagnosis of PV (87.4%; 95% CI, 81.5-91.9%) as compared to PF (69.0%; 95% CI, 49.2-84.7%; P = 0.018). The specificity for the diagnosis of pemphigus was 93.5% (95% CI, 91.1-95.4%). Patients with false-positive results (n = 37) were followed for a median duration of 5.3 years contributing 280.8 person-years. Thirty patients (81.1%) were eventually (...) The Usefulness of Indirect Immunofluorescence in Pemphigus and the Natural History of Patients With Initial False-Positive Results: A Retrospective Cohort Study The specificity and the predictive values of indirect immunofluorescence (IIF) in real-life settings is yet to be firmly established. The natural history of patients with false-positive results has not been sufficiently elucidated. The primary aim of the current study is to evaluate the diagnostic value of IIF analysis on monkey

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2018 Frontiers in medicine

86. Investigation of the Predisposing Factor of Pemphigus and its clinical subtype through a Genome-wide association and next generation sequence analysis. (PubMed)

Investigation of the Predisposing Factor of Pemphigus and its clinical subtype through a Genome-wide association and next generation sequence analysis. Pemphigus is an autoimmune blistering disease with pemphigus vulgaris (PV) and foliaceus (PF) as the two major histological subtypes. Associations with HLA molecules have been suggested, but specific HLA risk variants as well as non-HLA risk variants remain to be discovered.We performed a two-stage genome-wide association study in the Chinese (...) understanding of the genetic basis of pemphigus susceptibility and may offer opportunities for risk prediction and preventive treatment for pemphigus, in particular for PV.© 2018 European Academy of Dermatology and Venereology.

2018 Journal of the European Academy of Dermatology and Venereology

87. Retrospective analysis of a single-center clinical experience toward development of curative treatment of 123 pemphigus patients with a long-term follow-up: efficacy and safety of the multidrug protocol combining intravenous immunoglobulin with the cytoto (PubMed)

saving, the high dose and prolonged courses required for disease control are associated with significant adverse effects, including death. Although introduction of rituximab (RTX) provided for a favorable outcome, the high relapse rate, that is, up to 80%, precludes successful use of RTX as a monotherapy. Intravenous immunoglobulin (IVIg) is being increasingly utilized as off-label therapy for a variety of autoimmune and inflammatory diseases, including PV and pemphigus foliaceus (PF).The goal (...) Retrospective analysis of a single-center clinical experience toward development of curative treatment of 123 pemphigus patients with a long-term follow-up: efficacy and safety of the multidrug protocol combining intravenous immunoglobulin with the cytoto Pemphigus vulgaris (PV) is a life-long IgG autoantibody-mediated blistering disease affecting the mucosal surfaces lined by the stratified epithelium (oral, nasal, genital) and sometimes also the skin. While corticosteroid treatment is life

2018 International Journal of Dermatology

88. Rituximab and Low Dose Oral Immune Modulating Treatment to Maintain a Sustained Response in Severe Pemphigus Patients. (PubMed)

Rituximab and Low Dose Oral Immune Modulating Treatment to Maintain a Sustained Response in Severe Pemphigus Patients. This pilot retrospective study examined the role of continuous low-dose maintenance immunomodulatory treatment (IMT) as an adjunct to rituximab (RTX) rescue therapy in severe pemphigus vulgaris (PV) and pemphigus foliaceus (PF) after a complete response (CR) to RTX was achieved.Ten severe pemphigus patients who received RTX rescue therapy were evaluated after achieving CR (...) propose an alternative treatment strategy utilizing RTX as a rescue agent in combination with long-term LRMD as a means to maintain a sustained long-term CR post-RTX therapy in severe pemphigus patients. This strategy could prevent disease flares and the need for additional RTX cycles and higher dosages of immunomodulatory therapies.

2018 Journal of Dermatological Treatment

89. Paraneoplastic autoimmune multiorgan syndrome (PAMS): Beyond the single phenotype of paraneoplastic pemphigus. (PubMed)

including plakins, cadherins, alpha-2-macroglobulin like 1 (A2ML1), BP180, plakophilin-3, and several neuromuscular antigens. Originally described as paraneoplastic pemphigus in 1990 due to some of its clinical and immunologic similarities to classic pemphigus (pemphigus vulgaris and pemphigus foliaceus), PAMS is a multiorganopathy with several distinct features from these classic forms of pemphigus. Epidemiologically, PAMS is associated with underlying neoplasia and has a differing HLA-II allele (...) Paraneoplastic autoimmune multiorgan syndrome (PAMS): Beyond the single phenotype of paraneoplastic pemphigus. Paraneoplastic autoimmune multiorgan syndrome (PAMS) is characterized by a heterogenous group of signs and symptoms including severe desquamative stomatitis, a polymorphous cutaneous eruption, humoral immunity against plakin proteins, contribution of cell-mediated autoimmunity and commonly a progressive respiratory failure. Autoantibodies in PAMS target a wide array of antigens

2018 Autoimmunity reviews

90. Pemphigus Foliaceus—Repeated Treatment With Rituximab 7 Years After Initial Response: A Case Report (PubMed)

Pemphigus Foliaceus—Repeated Treatment With Rituximab 7 Years After Initial Response: A Case Report Pemphigus foliaceus is an autoimmune skin disease mediated by autoantibodies directed against desmoglein-1 located in the upper epidermal layer. Rituximab, a monoclonal anit-CD20 antibody depleting b-cells, offers an effective treatment possibility for therapy-resistant pemphigus foliaceus. Here, we present the case of 55-year-old man who did not respond sufficiently to conventional treatment

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2018 Frontiers in medicine

91. The Significance of Scalp Involvement in Pemphigus: A Literature Review (PubMed)

with pemphigus. Follicular acantholysis is a characteristic histopathological feature of pemphigus lesions localized on the scalp. Trichoscopy may serve as a supplementary method in the diagnosis of pemphigus. This review summarizes the most recent data concerning scalp involvement in pemphigus vulgaris and pemphigus foliaceus. A systematic literature search was conducted in three medical databases: PubMed, Embase, and Web of Science. The analysis included literature data about desmoglein distribution (...) The Significance of Scalp Involvement in Pemphigus: A Literature Review Scalp is a unique location for pemphigus because of the abundance of desmogleins localized in hair follicles. Scalp involvement is observed in up to 60% of patients in the course of pemphigus. The lesions may occasionally lead to alopecia. Unforced removal of anagen hairs in a pull test is a sign of high disease activity. Direct immunofluorescence of plucked hair bulbs is considered a reliable diagnostic method in patients

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2018 BioMed research international

92. Large International Validation of ABSIS and PDAI Pemphigus Severity Scores. (PubMed)

in a large international study. Consecutive patients with newly diagnosed pemphigus were enrolled in 31 centers. Severity scores were recorded during a 24-month period by the same two blinded investigators. Serum was collected at each visit for ELISA measurement of anti-desmoglein antibodies. The intraclass correlation coefficient (ICC) and Spearman rank correlation coefficient were calculated. A total of 116 patients with pemphigus vulgaris (n = 84) or pemphigus foliaceus (n = 32) were included (...) Large International Validation of ABSIS and PDAI Pemphigus Severity Scores. The Pemphigus Disease Area Index (PDAI) and Autoimmune Bullous Skin Disorder Intensity-Score (ABSIS) scores have been proposed to provide an objective measure of pemphigus activity. These scores have been evaluated only on already treated patients mainly with mild to moderate activity. The objective was to assess the interrater reliability of ABSIS and PDAI scores and their correlation with other severity markers

2018 Journal of Investigative Dermatology

93. Subtype-Specific Inherited Predisposition to Pemphigus in Chinese Population. (PubMed)

Subtype-Specific Inherited Predisposition to Pemphigus in Chinese Population. Pemphigus is a group of rare life-threatening mucocutaneous autoimmune diseases, presenting mainly as two subtypes: pemphigus vulgaris (PV) and pemphigus foliaceus (PF). Inherited predispositions to pemphigus have long been speculated but they remain poorly understood.To identify common and specific nongenetic and genetic factors associated with pemphigus and its subtypes in the Chinese population.A genome-wide (...) association study (GWAS) was performed in 496 unrelated patients with pemphigus (including 365 with PV and 104 with PF) and 1105 controls without pemphigus.A sex preference was observed only in PV (57·5% female) and not in PF (47·1% female). For male patients only, the mean age at diagnosis was significantly lower for PV than for PF (P < 0·001). The strongest associated single-nucleotide polymorphisms are in the human leucocyte antigen (HLA) region: rs70993900 (PV; P = 1·5 × 10-45 ) and rs9469220 (PF; P

2018 British Journal of Dermatology

94. Clinical and immunological features of pemphigus relapse. (PubMed)

prednisolone was tapered to around 0·1 mg kg-1 . In mucocutaneous pemphigus vulgaris (mcPV), the initial doses (mean ± SD) of prednisolone were significantly lower in patients with relapse (0·78 ± 0·24 mg kg-1 ) than in those without relapse (1·01 ± 0·01 mg kg-1 ). At relapse, mcPV shifted to mucosal dominant PV (mPV; 40%), pemphigus foliaceus (PF) (20%) or 'other' (20%). In contrast, relapsing mPV and PF had the same clinical phenotypes as the initial phenotypes. Patients with both anti-desmoglein (Dsg)1 (...) Clinical and immunological features of pemphigus relapse. More than half of patients with pemphigus experience relapse during the disease course. The risk factors and clinical and immunological characteristics of relapse remain largely unclear.To elucidate the risk factors and clinical features of pemphigus relapse.We carried out a retrospective review of the clinical records of 42 cases of pemphigus at a single centre.Sixty-two per cent of patients experienced relapse, usually when oral

2018 British Journal of Dermatology

95. A Study of PRN1008 in Patients With Pemphigus

patients with newly diagnosed or relapsing moderate to severe pemphigus (pemphigus vulgaris [PV] or pemphigus foliaceus [PF]) will be enrolled in the trial worldwide. The trial will last 68 weeks (approximately 17 months) for each participant. Patients will be randomized at Day 1, using a 1:1 ratio to receive PRN1008 or placebo twice per day, and by relapsing/newly diagnosed disease history (newly diagnosed defined as within 6 months of Screening). Study Design Go to Layout table for study information (...) to severe pemphigus, newly diagnosed or relapsing pemphigus vulgaris (PD) or pemphigus foliaceus (PF); positive anti-dsg1 or 3 autoantibody titer Laboratory clearance with adequate hematologic, hepatic, and renal function, body mass index (BMI) >17.5 Female patients who are of reproductive potential must agree for the duration of active treatment to use an effective means of contraception Able to provide written informed consent Exclusion Criteria: Previous use of a Bruton's tyrosine kinase (BTK

2018 Clinical Trials

96. Brazilian pemphigus foliaceus anti-desmoglein 1 autoantibodies cross-react with sand fly salivary LJM11 antigen (PubMed)

Brazilian pemphigus foliaceus anti-desmoglein 1 autoantibodies cross-react with sand fly salivary LJM11 antigen The environmental factors that contribute to the development of autoimmune diseases are largely unknown. Endemic pemphigus foliaceus in humans, known as Fogo Selvagem (FS) in Brazil, is mediated by pathogenic IgG4 autoantibodies against desmoglein 1 (Dsg1). Clusters of FS overlap with those of leishmaniasis, a disease transmitted by sand fly (Lutzomyia longipalpis) bites

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2012 Journal of immunology (Baltimore, Md. : 1950)

97. IgG Autoantibody Response against Keratinocyte Cadherins in Endemic Pemphigus Foliaceus (Fogo Selvagem). (PubMed)

IgG Autoantibody Response against Keratinocyte Cadherins in Endemic Pemphigus Foliaceus (Fogo Selvagem). It is well established that autoantibodies against desmoglein 3 and desmoglein 1 (Dsg1) are relevant in the pathogenesis of pemphigus vulgaris and pemphigus foliaceus, including its endemic form fogo selvagem (FS). Isolated reports have shown that in certain patients with these diseases, autoantibodies against other desmosomal cadherins and E-cadherin may also be present. The goal

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2012 Journal of Investigative Dermatology

98. Clearance Efficacy of Autoantibodies in Double Filtration Plasmapheresis for Pemphigus Foliaceus. (PubMed)

Clearance Efficacy of Autoantibodies in Double Filtration Plasmapheresis for Pemphigus Foliaceus. 22948906 2013 09 24 2015 11 19 1651-2057 93 2 2013 Mar 27 Acta dermato-venereologica Acta Derm. Venereol. Clearance efficacy of autoantibodies in double filtration plasmapheresis for pemphigus foliaceus. 181-2 10.2340/00015555-1444 Kasuya Akira A Moriki Mutsumi M Tatsuno Kazuki K Hirakawa Satoshi S Tokura Yoshiki Y eng Case Reports Letter Sweden Acta Derm Venereol 0370310 0001-5555 0 Autoantibodies (...) 0 Biomarkers 0 DSG1 protein, human 0 Desmoglein 1 0 Immunoglobulin A 0 Immunoglobulin G 0 Immunoglobulin M IM Aged, 80 and over Autoantibodies blood Biomarkers blood Desmoglein 1 immunology Humans Immunoglobulin A blood Immunoglobulin G blood Immunoglobulin M blood Male Middle Aged Pemphigus blood immunology therapy Plasmapheresis adverse effects methods Treatment Outcome 2012 9 6 6 0 2012 9 6 6 0 2013 9 26 6 0 ppublish 22948906 10.2340/00015555-1444

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2012 Acta Dermato-Venereologica

99. Profile of Trypanosoma cruzi Reactivity in a Population at High Risk for Endemic Pemphigus Foliaceus (Fogo Selvagem). (PubMed)

Profile of Trypanosoma cruzi Reactivity in a Population at High Risk for Endemic Pemphigus Foliaceus (Fogo Selvagem). Fogo selvagem (FS) is an autoimmune bullous disease with pathogenic IgG autoantibodies recognizing desmoglein 1 (Dsg1), a desmosomal glycoprotein. In certain settlements of Brazil, a high prevalence of FS (3%) is reported, suggesting environmental factors as triggers of the autoimmune response. Healthy individuals from endemic areas recognize nonpathogenic epitopes of Dsg1

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2012 American Journal of Tropical Medicine & Hygiene

100. Ultrastructure of acantholysis in pemphigus foliaceus reexamined from the current perspective. (PubMed)

Ultrastructure of acantholysis in pemphigus foliaceus reexamined from the current perspective. Pemphigus foliaceus (PF) is a chronic cutaneous autoimmune blistering disease that is characterized by superficial blistering of the skin, and according to the current perspective is caused by autoantibodies directed against desmoglein (Dsg) 1.To examine early acantholysis in the skin of patients with PF at an ultrastructural level.Two Nikolsky-negative (N-), five Nikolsky-positive (N+) and two

2012 British Journal of Dermatology

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