Werth, Victoria P

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2012 - 20132
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  • Publication
    Pemphigus
    (2013-10-01) Stoopler, Eric T; Santoro, Frank A; Werth, Victoria P
    Pemphigus vulgaris and paraneoplastic pemphigus are two subtypes of pemphigus that involve the oral mucosa. These autoimmune blistering disorders have antibodies targeted against proteins of keratinocyte adhesion, thereby causing acantholysis. Clinical findings include oral erosions and flaccid cutaneous bullae and erosions. In addition to the clinical exam, diagnostic tests including tissue biopsy, direct and indirect immunofluorescence, and enzyme-linked immunosorbent assays (ELISA) help to establish a diagnosis. Further malignancy workup in patients with suspected paraneoplastic pemphigus is warranted. Retrospective uncontrolled studies suggest that immunosuppressive agents reduce mortality in pemphigus vulgaris and cohort uncontrolled studies of rituximab, a monoclonal antibody against CD20, suggest it is an effective treatment for refractory patients. Ongoing studies will define its role in early disease.
  • Publication
    Lenalidomide Therapy in Treatment Refractory Cutaneous Lupus Erythematosus: Histologic and Circulating Leukocyte Profile and Potential Risk of a Systemic Lupus Flare
    (2012-04-01) Rosenbach, Misha; Okawa, Joyce; Braunstein, Inbal; Goodman, Noah G; Kovarik, Carrie; Prak, Eline Luning; Werth, Victoria P; Shah, Asha; Krathen, Michael
    Background Lenalidomide is a thalidomide analogue that may serve as an adjunctive therapy for treatment refractory cutaneous lupus erythematosus (CLE). Objectives We evaluate the use of lenalidomide in CLE and describe the skin and circulating leukocyte profile of treatment refractory patients before and after treatment. Patients/Methods Five subjects were treated with lenalidomide in an unblinded open-label study. Immunohistochemistry of skin was performed for T-cell markers, glycosaminoglycans and CXCL10, an interferon (IFN)-inducible chemokine, before and after treatment. Immunophenotyping and measurement of IFN-inducible genes from peripheral blood mononuclear cells was also performed before and after treatment. Results Four subjects demonstrated clinical improvement of their skin, however one of these responders subsequently developed symptoms of systemic lupus erythematosus. Small changes in rare circulating leukocyte subsets, plasmacytoid dendritic cells and regulatory T-cells, were observed with treatment and may correlate with clinical response. Treatment was associated with increased circulating HLA-DR expression and decreased markers of IFN-mediated pathways, regardless of clinical response. Limitations Our results are limited by small sample size and the measurement of rare populations of circulating cell subsets. Conclusions Lenalidomide may have utility as therapy for severe, treatment refractory CLE. However, our preliminary data suggest that lenalidomide may activate T-cells and trigger systemic disease in some CLE patients. We also saw a unique histologic and circulating leukocyte phenotype in the nonresponding subject. Further characterization of the skin and circulating leukocyte profile of treatment refractory patients will improve our understanding of CLE.