A case of paraneoplastic pemphigus associated with Castleman disease reacting with multiple autoantigens, including the p200 protein

Oh, Seung Joon; Lee, Sangeun; Hashimoto, Takashi; Kim, Soo Chan

doi:10.1111/bjd.14293

Cited by 9 publications

(7 citation statements)

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“…Previous studies reported the following as CD-associated cutaneous disorders: PNP (13), lichenoid eruptions (14), nodular eruptions (8), maculopapular eruptions (8), KS (15), autoimmune bleeding disorders, POEMS syndrome (16), cutaneous necrotizing vasculitis, and plane xanthoma (8). PNP is the most well-known CD-associated cutaneous disorder (17)(18)(19)(20)(21)(22)(23)(24)(25)(26). UCD is one of the most common causes of PNP, along with non-Hodgkin lymphoma and other haematological neoplasms (27).…”

Section: Discussionmentioning

confidence: 99%

“…The patient with PNP in the present study had UCD, consistent with previous reports. Oh et al (21) and Maier et al (24) suggested that autoantibodies, such as p200 proteins, Dsg1, Dsg3, and BP180, are involved in the pathogenesis of PNP. Furthermore, Sherman et al (8) postulated that xanthoma and CD share pathological mechanisms, including enhanced tissue uptake and increased lipolysis caused by the interaction of triglyceride-rich lipoprotein antibodies in the IgG-low density lipoprotein (LDL) complex with macrophage scavenger receptors that results in the formation of xanthomas and deposition of antibody-lipoprotein complexes.…”

Section: Discussionmentioning

confidence: 99%

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Cutaneous Disorders Associated with Castleman’s Disease

Kim¹,

Han²,

Bang³

et al. 2019

Acta Derm Venerol

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This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta Acta Derm Venereol 2019; 99: 984-989 984 SIGNIFICANCECastleman's disease is a rare lymphoproliferative disorder, which is often accompanied by various cutaneous manifestations. However, Castleman's disease-related cutaneous manifestations have not yet been systematically reviewed. The aim of this study was to examine the clinical, histopathological and laboratory characteristics of cutaneous manifestations in Castleman's disease, through a retrospective review of medical records. The study aimed to provide a comprehensive understanding of Castleman's disease-associated cutaneous disorders by subclassifying cutaneous disorders according to their underlying pathomechanisms. The study highlights the role of dermatologists in an in-depth review of various skin disorders related to Castleman's disease.Castleman's disease is a rare disease of the lymph nodes and related tissues, presenting as angiofollicular or giant lymph node hyperplasia. Although various skin manifestations have been reported to occur in Castleman's disease, a comprehensive study of cutaneous disorders in Castleman's disease is lacking. Therefore, the aim of this study was to investigate Castleman's disease-associated cutaneous disorders. The medical records of 57 patients with Castleman's disease who visited our hospitals from January 2007 to May 2018 were analysed retrospectively. Patients were classified according to the presence of skin involvement. Plasma variant-type Castleman's disease and multicentric Castleman's disease were more commonly found in patients with Castleman's disease with a cutaneous disorder than in those without a cutaneous disorder. In addition, the skin disorders were classified according to pathomechanisms: immune complexrelated (paraneoplastic pemphigus, xanthogranulomas), cytokine-related (vasculitis-like lesion, cherry angioma, hyperpigmentation), and non-specific (pruritus). This study builds on previous case reports of cutaneous disorders in Castleman's disease and proposes a new classification system.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Cutaneous Disorders Associated with Castleman’s Disease

Kim¹,

Han²,

Bang³

et al. 2019

Acta Derm Venerol

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“…32 gets and the development of other techniques to identify the presence of autoantibodies reinforces the need to update the diagnostic criteria 22,61. Joly et al determined that the occurrence of lymphoproliferative disease, positive IIF with rat bladder, and presence of anti-periplakin and anti-envoplakin with immunoprecipitation or immunoblotting are the most sensitive and specific findings for the diagnosis of PNP 51.…”

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confidence: 99%

Paraneoplastic pemphigus: a clinical, laboratorial, and therapeutic overview

Maruta

Miyamoto

Aoki

et al. 2019

An. Bras. Dermatol.

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Paraneoplastic pemphigus is a rare and severe autoimmune blistering disease characterized by mucocutaneous lesions associated with benign and malignant neoplasms. Diagnostic criteria include the presence of chronic mucositis and polymorphic cutaneous lesions with occult or confirmed neoplasia; histopathological analysis exhibiting intraepidermal acantholysis, necrotic keratinocytes, and vacuolar interface dermatitis; direct immunofluorescence with intercellular deposits (IgG and C3) and at the basement membrane zone (IgG); indirect immunofluorescence with intercellular deposition of IgG (substrates: monkey esophagus and simple, columnar, and transitional epithelium); and, autoreactivity to desmogleins 1 and 3, desmocollins 1, 2, and 3, desmoplakins I and II, envoplakin, periplakin, epiplakin, plectin, BP230, and α-2-macroglobulin-like protein 1. Neoplasias frequently related to paraneoplastic pemphigus include chronic lymphocytic leukemia, non-Hodgkin lymphoma, carcinomas, Castleman disease, thymoma, and others. Currently, there is no standardized treatment for paraneoplastic pemphigus. Systemic corticosteroids, azathioprine, mycophenolate mofetil, cyclosporine, rituximab, cyclophosphamide, plasmapheresis, and intravenous immunoglobulin have been used, with variable outcomes. Reported survival rates in 1, 2, and 5 years are 49%, 41%, and 38%, respectively.

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“…Second, antibodies against the 200-kDa laminin gamma-1 were identified. This is the second case of PNP with anti-laminin gamma-1 antibodies8. The role of these antibodies is yet to be described.…”

Section: Discussionmentioning

confidence: 83%

Paraneoplastic Pemphigus Associated with a Malignant Thymoma: A Case of Persistent and Refractory Oral Ulcerations Following Thymectomy

et al. 2017

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Paraneoplastic pemphigus is a rare, life-threatening autoimmune mucocutaneous blistering disease associated with underlying neoplasia, commonly lymphoproliferative tumors. Herein we report a case of paraneoplastic pemphigus with a unique autoantibody profile associated with a malignant thymoma. A 56-year-old female patient presented with relapsing oral ulcerations accompanied by erythematous papules and patches on her extremities for 2 months. Skin and mucosal biopsies identified interface dermatitis with lichenoid lymphocytic infiltration in the upper dermis. Immunoblotting and enzyme-linked immunosorbent assays revealed that the patient had multiple autoantibodies against desmoglein 1, desmocollin 1, 2, 3, laminin gamma-1, envoplakin, and periplakin. The skin lesions completely healed following thymectomy and systemic corticosteroid therapy, but the oral ulcerations persisted through a follow-up period of over 2 years.

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A case of paraneoplastic pemphigus associated with Castleman disease reacting with multiple autoantigens, including the p200 protein

Cited by 9 publications

References 0 publications

Cutaneous Disorders Associated with Castleman’s Disease

Cutaneous Disorders Associated with Castleman’s Disease

Paraneoplastic pemphigus: a clinical, laboratorial, and therapeutic overview

Paraneoplastic Pemphigus Associated with a Malignant Thymoma: A Case of Persistent and Refractory Oral Ulcerations Following Thymectomy

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