Idiopathic Multicentric Castleman Disease with Autoimmune Hemolytic Anemia and Production of Anti-drug Antibody against Tocilizumab

Tabata, Sakiko; Higuchi, Tomoaki; Tatsukawa, Seishiro; Narimatsu, Kazuyuki; Takeo, Hiroaki; Matsukuma, Susumu; Ito, Tsuyoshi

doi:10.2169/internalmedicine.2989-19

Cited by 9 publications

(9 citation statements)

References 18 publications

(20 reference statements)

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“…Although MCD was found to be the form of CD, most frequently accompanied by symptoms and signs of autoimmunity in previous reports, and iMCD has a close association with several autoimmune and autoinflammatory diseases, UCD with localized lesions and less systematic syndromes could also be occasionally complicated with diverse autoimmune disorders (Muskardin et al 2012 ; Fajgenbaum et al 2014 ; Liu et al 2016 ). Consistent with previous reports, AIHA and SS occurred in cases with PC variant, and MG in HV variant in this study (Yuzuriha et al 2011 ; Tabata et al 2019 ; Dei-Adomakoh et al 2018 ; Ishikawa et al 2014 ). In addition, the majority of PNP in our study was associated with HV type UCD as reported (Nikolskaia et al 2003 ).…”

Section: Discussionsupporting

confidence: 93%

“…Castleman disease (CD) represents a group of disorders characterized by polyclonal B lymphocyte proliferation, often associated with autoimmune manifestations, and occasionally be complicated with autoimmune connective tissue disease, such as systemic lupus erythematous (SLE), rheumatoid arthritis (RA), Sjogren’s syndrome (SS), mixed connective tissue disease, autoimmune hemolytic anemia (AIHA), myasthenia gravis (MG), amyloidosis, or thrombotic thrombocytopenic purpura (Muskardin et al 2012 ; Wang et al 2018 ; Ben-Chetrit et al 1989 ; Lee et al 2012 ; Yuzuriha et al 2011 ; Tabata et al 2019 ; Dei-Adomakoh et al 2018 ; Ishikawa et al 2014 ; Ogita et al 2007 ; London et al 2017 ). In addition, CD is related to the pathogenesis of paraneoplastic pemphigus (PNP) which is characterized by autoimmune-mediated cutaneous lesions with concomitant occurrence of either occult or confirmed systemic neoplasm (Bin et al 2019 ).…”

Section: Introductionmentioning

confidence: 99%

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Clinical characteristics and immunological abnormalities of Castleman disease complicated with autoimmune diseases

Sun

Chen²,

Wang

et al. 2021

J Cancer Res Clin Oncol

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Purpose To explore the clinical features and immunological mechanisms of Castleman disease (CD) complicated with autoimmune diseases (AID). Methods We explored the prevalence and clinical manifestations of CD complicated with AID by reviewing clinical, pathological, and laboratory data of 40 CD patients retrospectively, and then explored abnormal immune mechanisms in the co-existence of the two entities by monitoring lymphocyte subsets in peripheral blood. Results Paraneoplastic pemphigus, autoimmune hemolytic anemia, Sjogren’s syndrome, myasthenia gravis, and psoriasis were found to be coexisted with CD in 9/40 (22.5%) patients with different sequence of onset. No bias in the clinical and histological type of CD was observed for the occurrence of AID. CD patients with AID were more likely to have skin and/or mucous membrane damage and pulmonary complications, and presented elevated erythrocyte sedimentation rate, hypergammaglobulinemia, and positive autoantibodies than those without AID (p < 0.05). Deregulated cellular and innate immune responses as indicated by decreased CD3+ T cells and increased natural killer cells were observed in peripheral blood of CD patients with AID (p < 0.05). UCD patients with AID were successfully treated with surgery and immunosuppressive therapy. MCD complicated by AID relieved with immunosuppressors, cytotoxic chemotherapy, and rituximab. Conclusion Systemic inflammation/immunological abnormalities and organ dysfunction were associated with the occurrence of AID in CD. Impairment of cellular and innate immunity may be a candidate etiology for the coexistence of the two entities.

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Section: Discussionsupporting

confidence: 93%

Section: Introductionmentioning

confidence: 99%

Clinical characteristics and immunological abnormalities of Castleman disease complicated with autoimmune diseases

Sun

Chen²,

Wang

et al. 2021

J Cancer Res Clin Oncol

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“…In the largest series, anaemia was reported in 79 of 91 patients in whom haemoglobin was measured [ 12 ]. It is often microcytic and consistent with anaemia of chronic inflammation [ 1 , 26 ]. However, autoimmune haemolytic anaemia (AIHA) is a relatively frequent complication of MCD.…”

Section: Autoimmune Manifestations and Conditions Associated To Imcdmentioning

confidence: 99%

Idiopathic multicentric Castleman disease and associated autoimmune and autoinflammatory conditions: practical guidance for diagnosis

2022

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Idiopathic multicentric Castleman disease (iMCD) is an infrequent and life-threatening disorder characterised by systemic inflammatory symptoms, generalised lymphadenopathy, polyclonal lymphocyte proliferation, and organ dysfunction caused by a hyperinflammatory state. It accounts for one third to one half of all multicentric Castleman disease (MCD) cases. iMCD is often associated with autoimmune manifestations that may precede the iMCD diagnosis, be identified at the same time or follow it. In addition, iMCD may also coincide with a number of autoimmune diseases (such as psoriasis or myasthenia gravis) or autoinflammatory diseases (such as familial Mediterranean fever). Moreover, diverse inflammatory disorders, such as rheumatoid arthritis, systemic lupus erythematosus, adult-onset Still disease, systemic juvenile idiopathic arthritis, immunoglobulin (IgG4) related disease, or the recently described VEXAS syndrome, can present clinical features or lymphadenopathy with histopathological “Castleman-like” findings compatible with those of iMCD. Given the iMCD clinical heterogeneity and the overlap with other autoimmune or autoinflammatory disorders, iMCD diagnosis can be challenging. In this review, we explore the overlap between iMCD and inflammatory diseases and provide practical guidance on iMCD diagnosis in order to avoid misdiagnosis and confusion with other autoimmune or autoinflammatory conditions.

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“…However, the occurrence of autoimmune hemolytic anemia is rare, and six cases have been described to date, as far as the authors of this report are aware. [10][11][12][13][14][15] Interleukin-6 and Tocilizumab IL-6 is a pro-inflammatory cytokine that induces the differentiation and proliferation of T and B lymphocytes, is involved in the synthesis of acute phase proteins, in the stimulation of hematopoiesis and hepcidin production and in the development of constitutional symptoms present in various inflammatory diseases. In CD, hyperplastic lymph nodes with infiltration of plasma cells have a constitutionally increased production of IL-6.…”

Section: Etiologymentioning

confidence: 99%

Autoimmune hemolytic anemia in multicentric Castleman's disease: case report

da-Silva¹,

Alves²,

Cardinalli³

et al. 2022

AAAI

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A doença de Castleman é um distúrbio linfoproliferativo raro, podendo se manifestar sob a forma de massas localizadas ou como doença multicêntrica. A doença de Castleman multicêntrica é caracterizada por adenopatias generalizadas, visceromegalias, manifestações autoimunes e infecções recorrentes. Este artigo apresenta o relato de caso de anemia hemolítica autoimune por anticorpos quentes em paciente com doença de Castleman multicêntrica. Resposta eficaz foi obtida com uso de corticoterapia sistêmica e tocilizumabe.Descritores: Hiperplasia do linfonodo gigante, anemia hemolítica autoimune, anticorpos monoclonais.Castleman disease is a rare lymphoproliferative disorder that can manifest as localized masses or as multicentric disease. Multicentric Castleman disease is characterized by generalized adenopathies, visceromegaly, autoimmune manifestations, and recurrent infections. This article presents the case report of a patient with multicentric Castleman's disease and autoimmune hemolytic anemia by warm antibodies. Effective response was obtained with systemic corticotherapy and tocilizumab.

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Idiopathic Multicentric Castleman Disease with Autoimmune Hemolytic Anemia and Production of Anti-drug Antibody against Tocilizumab

Cited by 9 publications

References 18 publications

Clinical characteristics and immunological abnormalities of Castleman disease complicated with autoimmune diseases

Clinical characteristics and immunological abnormalities of Castleman disease complicated with autoimmune diseases

Idiopathic multicentric Castleman disease and associated autoimmune and autoinflammatory conditions: practical guidance for diagnosis

Autoimmune hemolytic anemia in multicentric Castleman's disease: case report

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