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Paraneoplastic Pemphigus

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181. Paraneoplastic pemphigus in association with hepatocellular carcinoma. (PubMed)

Paraneoplastic pemphigus in association with hepatocellular carcinoma. Paraneoplastic pemphigus (PNP) is an autoimmune mucocutaneous blistering disease associated with neoplasms, most frequently of the lymphoproliferative type. Rare PNP cases related to nonhematological solid tumors have been reported. The patient in this report presented with severe mucocutaneous involvement of PNP associated with hepatocellular carcinoma. Histopathology showed vacuolar interface dermatitis with keratinocyte (...) necrosis and intraepidermal acantholysis. Direct immunofluorescence exhibited deposition of intercellular IgG and complement and granular complement at the dermoepidermal junction. Indirect immunofluorescence testing showed a typical intercellular staining on monkey esophagus and rat bladder epithelium. Immunoprecipitation showed characteristic target antigens of 250, 210, and 190 kDa molecular weights. This patient met all diagnostic criteria for paraneoplastic pemphigus and is, to our knowledge

2003 Journal of American Academy of Dermatology

182. Progressive respiratory failure in paraneoplastic pemphigus associated with chronic lymphocytic leukemia. (PubMed)

Progressive respiratory failure in paraneoplastic pemphigus associated with chronic lymphocytic leukemia. A 70-year-old Caucasian man with chronic lymphocytic leukemia suffered from widespread, histologically proven cutaneous lichen planus responding to topical corticosteroids. 2 years later, he presented with painful erosive stomatitis and increasing dyspnea. Histology, direct and indirect immunofluorescence were diagnostic for paraneoplastic pemphigus. A full diagnostic workup could (...) not disclose the cause of the progressive respiratory insufficiency. Despite aggressive treatment of the lymphocytic leukemia and the paraneoplastic pemphigus, the patient died 3 months after diagnosis. Paraneoplastic pemphigus may lead to pulmonary failure which is refractory to treatment and has a fatal outcome.Copyright 2004 S. Karger AG, Basel

2004 Dermatology

183. Autoantibody production from a thymoma and a follicular dendritic cell sarcoma associated with paraneoplastic pemphigus. (PubMed)

Autoantibody production from a thymoma and a follicular dendritic cell sarcoma associated with paraneoplastic pemphigus. Paraneoplastic pemphigus (PNP) is an autoimmune mucocutaneous disease. We previously reported that B cells in a Castleman tumour associated with PNP produced autoantibodies. However, it is uncertain whether the production of autoantibodies from the associated tumour is a common mechanism in PNP.To investigate autoantibody production in a thymoma and a follicular dendritic

2005 British Journal of Dermatology

184. Paraneoplastic pemphigus associated with non-Hodgkin B-cell lymphoma and good response to prednisone. (Full text)

Paraneoplastic pemphigus associated with non-Hodgkin B-cell lymphoma and good response to prednisone. Paraneoplastic pemphigus is a life-threatening autoimmune bullous disease associated with neoplasia, generally of lymphoid origin. Immunosuppressive therapy is often disappointing and there are only a few reports of patients surviving more than 2 years. These cases were generally associated with benign neoplasms. We report here the case of a patient with paraneoplastic pemphigus associated

2005 Acta Dermato-Venereologica PubMed

185. Paraneoplastic pemphigus with clinical features of lichen planus associated with low-grade B cell lymphoma. (Full text)

Paraneoplastic pemphigus with clinical features of lichen planus associated with low-grade B cell lymphoma. Neoplasia-induced lichen planus is described as a cell-mediated reaction to unknown epithelial antigens. Paraneoplastic pemphigus (PNP), characterized by the presence of a specific array of autoantibodies, probably represents a different form of presentation of the same autoimmune syndrome where the mucocutaneous expression depends on the dominant pathologic mechanism.The authors report

2005 International Journal of Dermatology PubMed

186. Exacerbation of paraneoplastic pemphigus by cyclophosphamide treatment: detection of novel autoantigens and bronchial autoantibodies. (PubMed)

Exacerbation of paraneoplastic pemphigus by cyclophosphamide treatment: detection of novel autoantigens and bronchial autoantibodies. A 48-year-old woman with a follicular, grade III, B-cell non-Hodgkin lymphoma developed clinical, immunopathological and histological features of paraneoplastic pemphigus. The skin symptoms flared after repeated cyclophosphamide infusions, and were preceded and accompanied by a progressive dyspnoea. Although the skin and oral mucosal disease went into remission (...) serum contained IgG and IgA antibodies to desmocollin 3 as well as IgG antibodies to desmocollin 2. Serum IgG and IgA strongly stained rat bronchial epithelium, corresponding to autoantibodies possibly involved in the pathomechanism of the severe lung disease. In this case, which was characterized by a mixed IgA/IgG antibody panel displaying known and unique antigenicity, the serious episodes of paraneoplastic pemphigus flared after cyclophosphamide treatment.

2004 British Journal of Dermatology

187. Lichenoid paraneoplastic pemphigus in the absence of detectable antibodies. (PubMed)

Lichenoid paraneoplastic pemphigus in the absence of detectable antibodies. Paraneoplastic pemphigus (PNP) has been described as an antibody-mediated mucocutaneous disease occurring almost exclusively in patients with lymphocytic neoplasms. We describe 4 patients with the clinical features of the lichenoid variant of PNP in the absence of detectable autoantibodies. On the basis of these findings, we conclude that the spectrum of PNP likely includes patients with disease predominantly

2007 Journal of American Academy of Dermatology

188. Paraneoplastic pemphigus associated with CD20-positive follicular non-Hodgkin's lymphoma treated with rituximab: a third case resistant to rituximab therapy. (PubMed)

Paraneoplastic pemphigus associated with CD20-positive follicular non-Hodgkin's lymphoma treated with rituximab: a third case resistant to rituximab therapy. Paraneoplastic pemphigus is an IgG-mediated disease characterized clinically by a polymorphous blistering eruption with severe mucosal involvement associated with an underlying or occult malignancy. It is associated with high mortality, and response to treatment is generally poor. Potent immunosuppression is often necessary to control (...) progression of the disease. Three case reports have documented successful treatment of paraneoplastic pemphigus with rituximab, an anti-CD20 monoclonal antibody. However, two previous reports have noted that rituximab was unsuccessful in halting progression of PNP. We report a third case of paraneoplastic pemphigus associated with follicular non-Hodgkin's lymphoma in which rituximab was not effective. Whether rituximab is an effective and novel treatment for paraneoplastic pemphigus remains undecided.

2007 Clinical & Experimental Dermatology

189. Use of biosynthetic dressings in paraneoplastic pemphigus. (PubMed)

Use of biosynthetic dressings in paraneoplastic pemphigus. A 77-year-old woman with paraneoplastic pemphigus and non-Hodgkin's lymphoma was treated with supportive therapy and oral prednisone. Biobrane, a biosynthetic dressing, was later applied to the extensive areas of erosion to assist in pain management and to provide a temporary barrier function. She reported an improvement in the pain associated with the areas of erosion. The use of biosynthetic dressings in blistering disorders has

2004 Australasian Journal of Dermatology

190. Paraneoplastic pemphigus: a brief update. (PubMed)

Paraneoplastic pemphigus: a brief update. First described in 1990, paraneoplastic pemphigus is an autoimmune condition that causes considerable morbidity, is resistant to therapy and is frequently fatal. Clinical heterogeneity is being recognized as more cases are reported and the documented auto-antigen profile is also increasing. Target antigens are now known to be not restricted to the skin, suggesting that this condition is part of a paraneoplastic autoimmune multiorgan syndrome.

2005 Australasian Journal of Dermatology

191. A novel case of IgA paraneoplastic pemphigus associated with chronic lymphocytic leukemia. (PubMed)

A novel case of IgA paraneoplastic pemphigus associated with chronic lymphocytic leukemia. Paraneoplastic pemphigus is an autoimmune vesiculobullous and erosive mucocutaneous disease associated with an underlying malignancy. Reported malignancies include chronic lymphocytic leukemia, non-Hodgkin's lymphoma, Castleman's disease, sarcomas, and rarely solid tumors. Patients with paraneoplastic pemphigus develop characteristic IgG autoantibodies against several antigens including members (...) of the plakin family, bullous pemphigoid antigen 1, and desmosomal proteins. IgA pemphigus is another recently characterized immunobullous disease that presents as a vesiculopustular eruption with neutrophilic infiltration and epidermal acantholysis. Mucous membrane involvement is rare. We report what is to our knowledge a unique case with features of both IgA pemphigus and paraneoplastic pemphigus associated with chronic lymphocytic leukemia.

2007 Journal of American Academy of Dermatology

192. Paraneoplastic pemphigus without an underlying neoplasm. (PubMed)

Paraneoplastic pemphigus without an underlying neoplasm. We describe a 52-year-old man with paraneoplastic pemphigus (PNP) without any evidence of an underlying neoplasm over an 8-year follow-up period. He had a chronic relapsing vesiculobullous eruption for approximately 7 years (from April 1998 to May 2005). Initially, scattered flaccid vesicles with crusts developed on the face and trunk, which waxed and waned several times. Our patient was diagnosed as having PNP based on immunopathological

2007 British Journal of Dermatology

193. Paraneoplastic pemphigus associated with uterine carcinoma. (PubMed)

Paraneoplastic pemphigus associated with uterine carcinoma. Paraneoplastic pemphigus (PNP) is an autoimmune blistering disease characterized by severe mucous membrane involvement, polymorphous skin eruptions, and underlying neoplasms, usually those of lymphoreticular system. Cases of PNP associated with solid cancer are extremely rare. A 46-year-old woman presented with severe stomatitis, pseudomembranous conjunctivitis, and polymorphous skin eruptions. Histologic, immunofluorescence

2003 Journal of American Academy of Dermatology

194. Paraneoplastic pemphigus in association with Castleman's disease. (PubMed)

Paraneoplastic pemphigus in association with Castleman's disease. Paraneoplastic pemphigus (PNP) is an autoimmune mucocutaneous disease associated with lymphoproliferative neoplasms, and frequently with a very rare tumour, Castleman's disease.To analyse the clinical history, immunopathological and histopathological findings in 28 patients with a confirmed diagnosis of PNP and Castleman's disease.Sera from all patients were assayed by indirect immunofluorescence (IF) and immunoprecipitation (IP

2003 British Journal of Dermatology

195. Antibody titers to desmogleins 1 and 3 in a patient with paraneoplastic pemphigus associated with follicular dendritic cell sarcoma. (Full text)

Antibody titers to desmogleins 1 and 3 in a patient with paraneoplastic pemphigus associated with follicular dendritic cell sarcoma. Most paraneoplastic pemphigus (PNP) cases reported to date have been associated with lymphoproliferative neoplasms. Patients with PNP have autoantibodies against the plakin family (eg, envoplakin and periplakin). Antibodies against desmoglein 3 (Dsg3) and Dsg1, antigens for classic types of pemphigus, have also been reported to play an important role (...) the clinical phenotype and the anti-Dsg antibody profile, as seen in pemphigus vulgaris.

2004 Archives of Dermatology PubMed

196. Paraneoplastic pemphigus resembling linear IgA bullous dermatosis. (PubMed)

Paraneoplastic pemphigus resembling linear IgA bullous dermatosis. A 69-year-old Chinese man presented in 2001 with a blistering eruption over the upper and lower limbs associated with oral ulceration for 1 month. He had stage IIIA follicular small cell cleaved non-Hodgkin's lymphoma diagnosed 5 years previously, and had received several lines of palliative chemotherapy, including two courses of chlorambucil, six cycles of cyclophosphamide, adriamycin, vincristine, and prednisolone (CHOP (...) -intercellular antibody at a titre of 1/160 and positive on rat bladder at a titre of 1/80. A presumptive diagnosis of paraneoplastic pemphigus was made. This was later confirmed by the presence of antibodies against envoplakin (210 kDa), periplakin (190 kDa), and desmoglein 1 on immunoprecipitation studies. He was started on prednisolone 60 mg/day (1 mg/kg/day), with complete resolution of skin lesions within 1 week, but persistence of oral ulcers. Cyclophosphamide was added at a low dose of 1 mg/kg/day

2006 International Journal of Dermatology

197. Epitopes in the linker subdomain region of envoplakin recognized by autoantibodies in paraneoplastic pemphigus patients. (Full text)

Epitopes in the linker subdomain region of envoplakin recognized by autoantibodies in paraneoplastic pemphigus patients. Sera from paraneoplastic pemphigus (PNP) immunoprecipitate multiple antigens from human epidermal protein extract. In this study, we further characterized the autoantibodies in 12 PNP sera. Immunoblotting using recombinant linker subdomains of envoplakin, periplakin, desmoplakin, and bullous pemphigoid antigen I found that 11 of the 12 sera recognized linker subdomains

2006 Journal of Investigative Dermatology PubMed

198. Paraneoplastic autoimmune multiorgan syndrome (paraneoplastic pemphigus) in a child: case report and review of the literature. (Full text)

Paraneoplastic autoimmune multiorgan syndrome (paraneoplastic pemphigus) in a child: case report and review of the literature. 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

2004 Pediatrics PubMed

199. Human autoantibodies against desmoplakins in paraneoplastic pemphigus. (Full text)

Human autoantibodies against desmoplakins in paraneoplastic pemphigus. Recently, a previously unrecognized autoantibody mediated blistering disease, paraneoplastic pemphigus has been described. Paraneoplastic pemphigus is associated with lymphoid malignancies, thymomas, and poorly differentiated sarcomas. Serum of affected patients contain pathogenic autoantibodies that immunoprecipitate from normal keratinocytes a characteristic complex of four polypeptides with M(r) of 250, 230, 210, and 190 (...) to homogeneity from pig tongue epithelium. By indirect immunofluorescence, 9 of 11 sera showed strong binding to epithelial and nonepithelial desmosomes, and 2 were weakly reactive. All 11 immunoprecipitated 250- and 210-kD bands of variable intensity that comigrated with bands identified by a murine monoclonal antidesmoplakin antibody, and immunoblotting confirmed binding of the serum autoantibodies to purified desmoplakins. This demonstrates that paraneoplastic pemphigus is the first human autoimmune

1992 Journal of Clinical Investigation PubMed

200. Paraneoplastic pemphigus in children and adolescents. (PubMed)

Paraneoplastic pemphigus in children and adolescents. Paraneoplastic pemphigus (PNP) is an autoimmune mucocutaneous disease associated with specific B-cell lymphoproliferative neoplasms. There has been an increasing number of individual reports in the childhood and adolescent population.To examine the clinical and immunopathological features of PNP occurring in children and adolescents.We analysed the clinical and immunopathological findings of 14 patients under the age of 18 years

2002 British Journal of Dermatology

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