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Grant J. Anhalt - One of the best experts on this subject based on the ideXlab platform.
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Paraneoplastic Pemphigus Associated with Pelvic Inflammatory Fibrosarcoma: A Case Report
The Journal of dermatology, 2005Co-Authors: Claudia Giuli Santi, Grant J. Anhalt, Rosalba Sánchez Flores, Martha Martínez Medina, Celina Wakisaka Maruta, Valeria Aoki, Evandro A. RivittiAbstract:A 36-year-old African-American woman presented with an extensive stomatitis and pigmented cutaneous macules on the neck, axillae and hands. Subsequently she developed violaceus papules on the dorsa of the hands, histologically consistent with an interface dermatitis. After 18 months of progressive disease, Paraneoplastic Pemphigus was suspected and a search for an underlying neoplasm was initiated. An exploratory laparotomy revealed a pelvic mass and the histologic examination showed an inflammatory fibrosarcoma. The evidence of acantholysis on new cutaneous lesions and the positivity of indirect immunofluorescence with rodent urinary bladder epithelium reinforced the diagnostic criteria for Paraneoplastic Pemphigus, which is confirmed by the identification of strong protein bands at 210, 190 and 170 kd by immunoprecipitation. Paraneoplastic Pemphigus should be considered when investigating atypical mucocutaneous manifestations of Pemphigus vulgaris and lichen planus. Diagnostic screening for Paraneoplastic Pemphigus and a search for an underlying tumor should be performed.
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CD8+ T lymphocytes in bronchiolitis obliterans, Paraneoplastic Pemphigus, and solitary Castleman's disease.
The New England journal of medicine, 2003Co-Authors: Mark A. Hoffman, Xiaoying Qiao, Grant J. AnhaltAbstract:To the Editor: Paraneoplastic Pemphigus has been reported in patients with Castleman's disease, and bronchiolitis obliterans in association with Paraneoplastic Pemphigus can eventuate in respirator...
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Lichenoid Dermatitis in Paraneoplastic Pemphigus A Pathogenic Trigger of Epitope Spreading
Archives of dermatology, 2000Co-Authors: Glen M. Bowen, Grant J. Anhalt, Kevin D. Cooper, Neill T. Peters, David P. Fivenson, Hossein C. Nousari, Seth R. StevensAbstract:Background In select cases, lichen planus has been observed to be a Paraneoplastic condition sometimes associated with Paraneoplastic Pemphigus, a disease featuring autoantibodies directed against plakin proteins, desmogleins 3 and 1, and a still uncharacterized 170-kd antigen. Epitope spreading describes the phenomenon where underlying chronic inflammation leads to the sequential recognition of new epitopes on self-proteins over time. Observations Five of 6 patients diagnosed as having Paraneoplastic Pemphigus had concomitant clinical and histological features of lichen planus. In 1 patient, results of the initial indirect immunofluorescence on rat bladder were negative and only 2 of the 5 antigens were identified by immunoprecipitation. After 1 year of worsening disease, repeated testing confirmed the presence of antibodies directed against all 6 of the implicated antigens, supportive of our hypothesis that epitope spreading may occur in Paraneoplastic Pemphigus. Conclusions Lichenoid eruptions may predispose to an early evolutionary stage of Paraneoplastic Pemphigus. Cell-mediated autoimmunity at the dermoepidermal junction may promote the exposure of self-antigens and the development of subsequent and progressive humoral autoimmunity. As such, Paraneoplastic Pemphigus may demonstrate epitope spreading in a human, humoral-mediated autoimmune disease.
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Paraneoplastic Pemphigus appearing as bullous pemphigoid like eruption after palliative radiation therapy
Journal of The American Academy of Dermatology, 1993Co-Authors: Richard G Fried, Yelva Lynfield, Patricia Vitale, Grant J. AnhaltAbstract:Paraneoplastic Pemphigus is a recently described disease in which patients have polymorphous skin lesions suggestive of both erythema multiforme major and Pemphigus vulgaris in association with internal neoplasms, especially non-Hodgkin's lymphoma. These patients have characteristic autoantibodies that bind specific epidermal proteins. A Nikolsky-negative bullous pemphigoid-like eruption developed within the radiation therapy field in a 72-year-old man receiving palliative treatment for recurrent large cell lymphoma. The eruption rapidly progressed to a Nikolsky-positive bullous process more typical of Pemphigus vulgaris with extensive involvement of respiratory epithelia. Despite aggressive treatment with high-dose corticosteroids and antibiotics, the patient rapidly succumbed. Results of immunofluorescence studies and autopsy findings confirmed the diagnosis of Paraneoplastic Pemphigus.
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Paraneoplastic Pemphigus Presenting as a Lichen Planus Pemphigoides—like Eruption
Archives of dermatology, 1993Co-Authors: Seth R. Stevens, Grant J. Anhalt, Christopher E.m. Griffiths, Kevin D. CooperAbstract:• Background.— Paraneoplastic Pemphigus refers to a specific disease entity in which neoplasia is associated with severe mucosal ulceration and polymorphous cutaneous eruptions, sometimes resembling erythema multiforme. Direct immunofluorescence and indirect immunofluorescence on standard substrates are similar to Pemphigus vulgaris; however, Paraneoplastic Pemphigus serum uniquely binds to all epithelia and has distinctive immunoprecipitation characteristics. Observations.— A 63-year-old woman with chronic lymphocytic leukemia developed a severe, mucocutaneous, vesiculobullous eruption with the clinical and histopathologic characteristics of lichen planus pemphigoides evolving into a Stevens-Johnson—like presentation and the immunofluorescence findings of Pemphigus vulgaris. Evaluation of her serum confirmed the presence of autoantibodies specific for Paraneoplastic Pemphigus by indirect immunofluorescence and immunoprecipitation criteria. Conclusion.— The spectrum of Paraneoplastic Pemphigus must now be expanded to include cases that present clinically and histologically as a lichen planus pemphigoides— like eruption. ( Arch Dermatol. 1993;129:866-869)
W F Bergfeld - One of the best experts on this subject based on the ideXlab platform.
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Indirect immunofluorescence on rat bladder transitional epithelium: a test with high specificity for Paraneoplastic Pemphigus.
Journal of the American Academy of Dermatology, 1993Co-Authors: A Y Liu, R Valenzuela, T N Helm, C Camisa, A L Melton, W F BergfeldAbstract:Paraneoplastic Pemphigus is a blistering disease with specific serum immunoprecipitation findings. Although immunoprecipitation studies allow accurate diagnosis, they are time-consuming, expensive, and not readily available. In contrast, indirect immunofluorescence (IIF) testing of serum on transitional rat bladder epithelium is a simple and inexpensive method available to any immunopathology laboratory. Our purpose was to determine the specificity of positive IIF on rat bladder epithelium for Paraneoplastic Pemphigus. The IIF findings in four index cases of Paraneoplastic Pemphigus were compared with the findings in 47 patients with a variety of malignant neoplasms and no associated blistering disease as well as 49 patients with vesiculobullous or lichenoid disease but no neoplasia. IIF was negative in all patients with neoplasia and no blistering disease and negative in all but one of the patients with vesiculobullous or lichenoid disease without neoplasia (98.9% specificity). IIF on transitional rat bladder epithelium appears to be a highly specific test for Paraneoplastic Pemphigus. Because of its simplicity and inexpensiveness, we suggest that IIF be performed on transitional epithelium in any suspected case of Paraneoplastic Pemphigus.
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Indirect immunofluorescence on rat bladder transitional epithelium: a test with high specificity for Paraneoplastic Pemphigus.
Journal of the American Academy of Dermatology, 1993Co-Authors: A Y Liu, Charles Camisa, Rafael Valenzuela, Thomas N. Helm, A L Melton, W F BergfeldAbstract:Background: Paraneoplastic Pemphigus is a blistering disease with specific serum immuno-precipitation findings. Although immunoprecipitation studies allow accurate diagnosis, they are time-consuming, expensive, and not readily available. In contrast, indirect immunofluorescence (IIF) testing of serum on transitional rat bladder epithelium is a simple and inexpensive method available to any immunopathology laboratory. Objective: Our purpose was to determine the specificity of positive IIF on rat bladder epithelium for Paraneoplastic Pemphigus. Methods: The IIF findings in four index cases of Paraneoplastic Pemphigus were compared with the findings in 47 patients with a variety of malignant neoplasms and no associated blistering disease as well as 49 patients with vesiculobullous or lichenoid disease but no neoplasia. Results: IIF was negative in all patients with neoplasia and no blistering disease and negative in all but one of the patients with vesiculobullous or lichenoid disease without neoplasia (98.9% specificity). Conclusion: IIF on transitional rat bladder epithelium appears to be a highly specific test for Paraneoplastic Pemphigus. Because of its simplicity and inexpensiveness, we suggest that IIF be performed on transitional epithelium in any suspected case of Paraneoplastic Pemphigus.
A Y Liu - One of the best experts on this subject based on the ideXlab platform.
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Indirect immunofluorescence on rat bladder transitional epithelium: a test with high specificity for Paraneoplastic Pemphigus.
Journal of the American Academy of Dermatology, 1993Co-Authors: A Y Liu, R Valenzuela, T N Helm, C Camisa, A L Melton, W F BergfeldAbstract:Paraneoplastic Pemphigus is a blistering disease with specific serum immunoprecipitation findings. Although immunoprecipitation studies allow accurate diagnosis, they are time-consuming, expensive, and not readily available. In contrast, indirect immunofluorescence (IIF) testing of serum on transitional rat bladder epithelium is a simple and inexpensive method available to any immunopathology laboratory. Our purpose was to determine the specificity of positive IIF on rat bladder epithelium for Paraneoplastic Pemphigus. The IIF findings in four index cases of Paraneoplastic Pemphigus were compared with the findings in 47 patients with a variety of malignant neoplasms and no associated blistering disease as well as 49 patients with vesiculobullous or lichenoid disease but no neoplasia. IIF was negative in all patients with neoplasia and no blistering disease and negative in all but one of the patients with vesiculobullous or lichenoid disease without neoplasia (98.9% specificity). IIF on transitional rat bladder epithelium appears to be a highly specific test for Paraneoplastic Pemphigus. Because of its simplicity and inexpensiveness, we suggest that IIF be performed on transitional epithelium in any suspected case of Paraneoplastic Pemphigus.
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Indirect immunofluorescence on rat bladder transitional epithelium: a test with high specificity for Paraneoplastic Pemphigus.
Journal of the American Academy of Dermatology, 1993Co-Authors: A Y Liu, Charles Camisa, Rafael Valenzuela, Thomas N. Helm, A L Melton, W F BergfeldAbstract:Background: Paraneoplastic Pemphigus is a blistering disease with specific serum immuno-precipitation findings. Although immunoprecipitation studies allow accurate diagnosis, they are time-consuming, expensive, and not readily available. In contrast, indirect immunofluorescence (IIF) testing of serum on transitional rat bladder epithelium is a simple and inexpensive method available to any immunopathology laboratory. Objective: Our purpose was to determine the specificity of positive IIF on rat bladder epithelium for Paraneoplastic Pemphigus. Methods: The IIF findings in four index cases of Paraneoplastic Pemphigus were compared with the findings in 47 patients with a variety of malignant neoplasms and no associated blistering disease as well as 49 patients with vesiculobullous or lichenoid disease but no neoplasia. Results: IIF was negative in all patients with neoplasia and no blistering disease and negative in all but one of the patients with vesiculobullous or lichenoid disease without neoplasia (98.9% specificity). Conclusion: IIF on transitional rat bladder epithelium appears to be a highly specific test for Paraneoplastic Pemphigus. Because of its simplicity and inexpensiveness, we suggest that IIF be performed on transitional epithelium in any suspected case of Paraneoplastic Pemphigus.
Charles Camisa - One of the best experts on this subject based on the ideXlab platform.
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Paraneoplastic Pemphigus occurring in a patient with B-cell non-Hodgkin's lymphoma.
Cutis, 1998Co-Authors: Todd E. Schlesinger, Charles Camisa, Karen Mccarron, Grant James AnhaltAbstract:We present the case of a 71-year-old white male with Paraneoplastic Pemphigus associated with a B-cell non-Hodgkin's lymphoma. Diagnosis of Paraneoplastic Pemphigus was made by the characteristic findings on immunoprecipitation performed on a serum specimen. Paraneoplastic Pemphigus is a severe autoimmune disease comprised of polymorphous mucocutaneous lesions, characteristic laboratory findings, association with one of several types of neoplasms, and a very poor prognosis.
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Paraneoplastic Pemphigus. A distinct autoimmune vesiculobullous disorder associated with neoplasia.
Oral surgery oral medicine and oral pathology, 1993Co-Authors: Thomas N. Helm, Charles Camisa, Rafael Valenzuela, Carl M. AllenAbstract:Abstract A vesiculobullous disease termed Paraneoplastic Pemphigus with distinct autoantibodies was newly described in 1990. All reported cases have occurred in patients with a history of neoplasia, including lymphoma, chronic lymphocytic leukemia, poorly differentiated sarcoma, and benign thymoma. As in Pemphigus vulgaris, intraepithelial clefts with acantholysis are noted hisopathologically, and intercellular binding of immunoreactants is seen with direct immunofluorescence studies of mucous membrane and skin biopsies. However, immunoreactants may also be found along the basement membrane zone in Paraneoplastic Pemphigus. Indirect immunofluorescence using rat bladder epithelium as substrate shows an intercellular pattern that appears to be highly specific for Paraneoplastic Pemphigus. We report a patient with non-Hodgkins lymphoma of 8 years duration who developed severe erosive stomatitis and lichenoid dermatitis after receiving chemotherapy for a relapse of lymphoma. Her case illustrates the typical features of the disorder described as Paraneoplastic Pemphigus. Neoplasia-associated Pemphigus may be a more precise term for this disorder because the course of the blistering eruption does not always parallel the course of the underlying cancer. The clinical features, histopathologic findings, and immunofluorescence findings of this unique syndrome are reviewed.
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Indirect immunofluorescence on rat bladder transitional epithelium: a test with high specificity for Paraneoplastic Pemphigus.
Journal of the American Academy of Dermatology, 1993Co-Authors: A Y Liu, Charles Camisa, Rafael Valenzuela, Thomas N. Helm, A L Melton, W F BergfeldAbstract:Background: Paraneoplastic Pemphigus is a blistering disease with specific serum immuno-precipitation findings. Although immunoprecipitation studies allow accurate diagnosis, they are time-consuming, expensive, and not readily available. In contrast, indirect immunofluorescence (IIF) testing of serum on transitional rat bladder epithelium is a simple and inexpensive method available to any immunopathology laboratory. Objective: Our purpose was to determine the specificity of positive IIF on rat bladder epithelium for Paraneoplastic Pemphigus. Methods: The IIF findings in four index cases of Paraneoplastic Pemphigus were compared with the findings in 47 patients with a variety of malignant neoplasms and no associated blistering disease as well as 49 patients with vesiculobullous or lichenoid disease but no neoplasia. Results: IIF was negative in all patients with neoplasia and no blistering disease and negative in all but one of the patients with vesiculobullous or lichenoid disease without neoplasia (98.9% specificity). Conclusion: IIF on transitional rat bladder epithelium appears to be a highly specific test for Paraneoplastic Pemphigus. Because of its simplicity and inexpensiveness, we suggest that IIF be performed on transitional epithelium in any suspected case of Paraneoplastic Pemphigus.
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Conjunctival Involvement in Paraneoplastic Pemphigus
American journal of ophthalmology, 1992Co-Authors: Stephanie J. Meyers, Charles Camisa, Gary A. Varley, David M. Meisler, Arden H. WanderAbstract:Paraneoplastic Pemphigus is a recently described autoimmune inflammatory mucocutaneous disease associated with an underlying neoplasm. Although histopathologic and direct immunofluorescence findings of involved skin and mucous membranes are consistent with Pemphigus vulgaris, indirect immunofluorescence and immunoprecipitation study results are unique. We treated two patients with non-Hodgkin's lymphoma and Paraneoplastic Pemphigus. Both patients had bilateral bulbar conjunctival hyperemia and diffuse papillary tarsal conjunctival reactions. One patient had sloughing of conjunctival epithelium and the other had tarsal conjunctival cicatrization and forniceal shortening. Histopathologic findings of conjunctivae obtained from both patients were consistent with Pemphigus vulgaris. Diffuse deposition of IgG and C3 in the intercellular substance of the conjunctival epithelium was demonstrated by direct immunofluorescence. Indirect immunofluorescence testing disclosed binding of autoantibodies to rodent bladder and intestinal epithelium. Immunoprecipitation disclosed antibodies reactive to Desmoplakin I (250 kd), bullous pemphigoid (230 kd), Desmoplakin II (210 kd) and 190-kd proteins. Ophthalmologists and pathologists should be aware of the conjunctival changes in Paraneoplastic Pemphigus.
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Perplexing Parlance of Paraneoplastic Pemphigus
JAMA, 1992Co-Authors: Thomas M. Helm, Charles Camisa, Rafael ValenzuelaAbstract:To the Editor. —We read with interest the report by Fullerton et al 1 of a patient with Paraneoplastic Pemphigus after autologous bone marrow transplantation. Of note, their patient had no evidence of diffuse large-cell lymphoma at the time of autopsy. His lymphoma seems to have been in remission for the 6 months prior to the development of his blistering cutaneous eruption. In the last sentence of the article, the authors write "our patient clearly demonstrates that an associated neoplasm need not be active or detectable" in Paraneoplastic Pemphigus. This brings up the question of whether Paraneoplastic Pemphigus is truly Paraneoplastic. The Greek prefix para - means beside, and the course of Paraneoplastic phenomena has traditionally been expected to approximately parallel a malignant process in order for a causal relationship to be demonstrated. 2 Fullerton et al elegantly demonstrate that their patient had the same disorder described by Anhalt et al.
Rita S. Jerath - One of the best experts on this subject based on the ideXlab platform.
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Paraneoplastic Autoimmune Multiorgan Syndrome (Paraneoplastic Pemphigus) in a Child: Case Report and Review of the Literature
Pediatrics, 2004Co-Authors: Joshua E. Lane, Carol M. Woody, Loretta S. Davis, Margaret Frank Guill, Rita S. JerathAbstract:Paraneoplastic autoimmune multiorgan syndrome, also known as Paraneoplastic Pemphigus, has been observed only rarely among children. We describe a 10-year-old boy with typical clinical and histologic findings of Paraneoplastic Pemphigus associated with Castleman's disease. His disease was refractory to resection of the tumor and aggressive combination immunosuppressive therapies. The patient died 1 year after presentation, as a result of complications of bronchiolitis obliterans. This case is unusual because of the young age of the patient.