Multicentric Castleman’s disease associated with IgA vasculitis (Henoch-Schönlein purpura) responding well to tocilizumab: a case report

Ōshima, Yōichi; Hoshino, Junichi; Suwabe, Tatsuya; Hayami, Noriko; Yamanouchi, Masayuki; Sekine, Akinari; Ueno, Toshiharu; Mizuno, Hiroki; Yabuuchi, Junko; Imafuku, Aya; Kawada, Manabu; Hiramatsu, Rikako; Hasegawa, Eiko; Sawa, Naoki; Takaichi, Kenmei; Hayashi, Nobuyuki; Fujii, Takeshi; Ubara, Yoshifumi

doi:10.1007/s10067-017-3568-y

Cited by 12 publications

(9 citation statements)

References 18 publications

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“…IL-6, IL-12, TGF-1ß, and TNF-α are the main cytokines proposed to drive POEMS symptoms (29). Oshima et al (30) suggested that the overproduction of IL-6 may contribute to vasculitis as well as the pathogenesis of CD. Furthermore, increased IL-6 in MCD causes B cell proliferation and VEGF secretion, which is critical to the pathogenesis of haemangiomas (i.e.…”

Section: Discussionmentioning

confidence: 99%

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%

Cutaneous Disorders Associated with Castleman’s Disease

Kim¹,

Han²,

Bang³

et al. 2019

Acta Derm Venerol

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“…At the same time, pathological analysis revealed that the major pathological type of UCD and MCD was the hyaline-vascular type and plasma-cell type, respectively. In this study, there were 8 cases of progressive enlargement of lymph nodes (4.0–7.0 cm in diameter), including 2 cases in retroperitoneal lymph nodes, 4 cases in cervical lymph nodes, and 2 cases in mediastinal lymph nodes, suggesting that the risk of enlargement of lymph nodes is higher in CD child patients, showing differences from adults with enlargement of abdominal and mediastinal lymph nodes [ 17 ]. There is a lack of significant lymphatic symptoms in the initial stage of the disease, but local compression can occur with the constant enlargement of lymph nodes, leading to different symptoms based on the different sites of compression.…”

Section: Discussionmentioning

confidence: 99%

Analysis of Clinical Characteristics, Pathological Changes and Changes of Interleukin-6 (IL-6) and C-Reactive Protein (CRP) in Children with Castleman’s Disease

Zou

Pan

et al. 2020

Med Sci Monit

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Background The aim of this study was to analyze the pathological changes, clinical characteristics and changes in immunity, interleukin-6 (IL-6) and C-reactive protein (CRP) in children with Castleman’s disease (CD). Material/Methods A total of 15 CD child patients were enrolled as observation group, while 20 normal children receiving healthy examination were enrolled as healthy control group. The pathological changes, clinical characteristics and changes in immunity and serum IL-6 and CRP expressions were retrospectively analyzed in observation group. Results The clinical manifestation of unicentric CD (UCD) was mainly enlargement of cervical lymph nodes without liver-spleen enlargement and fever, and the major pathological type was the hyaline-vascular type. Multicentric CD (MCD) child patients all had anemia, fever and other systemic symptoms, and the major pathological type was the plasma-cell type. There were expressions of the immune indexes, including cluster of differentiation 3 (CD3), CD4, CD8, CD20, and CD79, in a certain degree, while CD138 and VS38C expressions displayed the polyclonal proliferation of plasma cells, rather than neoplastic proliferation. The Epstein-Barr virus and human herpes virus-8 detection results were negative, and CD21 in follicular dendritic cells in abnormal germinal center was positive. The expression levels of serum IL-6 and CRP in observation group were higher than those in control group ( P <0.05). Conclusions There are immune dysfunction and increased expressions of serum IL-6 and CRP in child patients with CD, so it can be speculated that the immune abnormality and overexpression of serum inflammatory factors may be associated with the occurrence of CD.

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“…However, since there is no standard therapy, treatment recommendations for CD can be difficult [ 3 ]. Recently, TCZ was shown to be effective for treatment and has been widely used in Japan since June 2005 [ 1 , 2 , 9 , 10 ].…”

Section: Discussionmentioning

confidence: 99%

The efficacy and safety of anti-interleukin-6 receptor monoclonal blockade in a renal transplant patient with Castleman disease: early post-transplant outcome

et al. 2018

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BackgroundMulticentric Castleman disease (MCD) is an uncommon lymphoproliferative disease characterized by systemic inflammatory reactions associated with the dysregulated production of interleukin-6 (IL-6). In patients with MCD, renal involvement is uncommon, with only one report published regarding kidney transplantation (KTx) to treat end-stage renal disease (ESRD) secondary to MCD. Recent clinical observations have shown that IL-6 production is implicated in allograft rejection, while IL-6 receptor blockade (with tocilizumab [TCZ]) reduces alloantibody generation and thereby improves graft survival; however, the efficacy and safety of TCZ in MCD patients undergoing KTx is still unknown.Case presentationHerein, we describe the case of a 50-year-old man with MCD who received living-donor KTx for ESRD. Post-operative immunosuppression consisted of a triple-drug regimen (tacrolimus, mycophenolate mofetil and methylprednisolone) with TCZ that was administered intravenously every 2 weeks. At 17 months post-transplantation, the patient remains asymptomatic, and the allograft pathology has shown no evidence of rejection and no development of de novo donor-specific antibody (DSA).ConclusionsTo our knowledge, this is the second reported case of an MCD patient with ESRD who underwent successful KTx. TCZ safely supported the patient during the perioperative period, and this drug may be useful for blocking the generation of donor-specific antibodies and reducing the risk of rejection episodes. KTx in combination with TCZ is thus considered a viable treatment option for ESRD due to MCD.

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Multicentric Castleman’s disease associated with IgA vasculitis (Henoch-Schönlein purpura) responding well to tocilizumab: a case report

Cited by 12 publications

References 18 publications

Cutaneous Disorders Associated with Castleman’s Disease

Cutaneous Disorders Associated with Castleman’s Disease

Analysis of Clinical Characteristics, Pathological Changes and Changes of Interleukin-6 (IL-6) and C-Reactive Protein (CRP) in Children with Castleman’s Disease

The efficacy and safety of anti-interleukin-6 receptor monoclonal blockade in a renal transplant patient with Castleman disease: early post-transplant outcome

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