Multicentric Castleman disease mimicking IgG4-related disease: A case report

Izumi, Yuichi; Takeshita, Hayato; Moriwaki, Yuji; Hisatomi, Kazuki; Matsuda, Masakazu; Yamashita, Nami; Kawahara, Chieko; Shigemitsu, Yoshika; Iwanaga, Naoki; Kawakami, Atsushi; Kurohama, Hirokazu; Niino, Daisuke; Ito, Masahiro; Migita, Kiyoshi

doi:10.3109/14397595.2014.985356

Cited by 11 publications

(7 citation statements)

References 20 publications

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“…Whether cardiovascular patients with IgG4-positive cell infiltration have a different clinical course [ 23 ] and an altered responsiveness to drug therapy as compared with those without IgG4-positive cell infiltration should be analyzed in future longitudinal studies. Third, IgG4-positive lymphoplasmacytic infiltration is not a feature exclusive to IgG4-RD; it may be observed in several immune and/or inflammatory disorders such as Castleman disease [ 24 ], Rosai-Dorfman disease [ 25 ], Wegener granulomatosis [ 26 ], and sialadenitis caused by sialolithiasis [ 27 ]. Lastly, we examined the presence or absence of IgG4-positive cell infiltration in one slide for each of the 103 samples; therefore, the true prevalence of IgG4-positive cell infiltration among these surgical samples might have been greater than what we have observed.…”

Section: Discussionmentioning

confidence: 99%

IgG4-positive cell infiltration in various cardiovascular disorders - results from histopathological analysis of surgical samples

Hourai

Kasashima

Sohmiya

et al. 2017

BMC Cardiovasc Disord

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BackgroundThe diagnosis of Immunoglobulin G4 (IgG4)-related disease (IgG4-RD), in general, depends on serum IgG4 concentrations and histopathological findings; therefore, diagnosis of IgG4-RD in cardiovascular organs/tissues is often difficult owing to the risk of tissue sampling.MethodsPrevalence of IgG4-positive lymphoplasmacytic infiltration in 103 consecutive cardiovascular surgical samples from 98 patients with various cardiovascular diseases was analyzed immunohistochemically.ResultsThe diagnoses of the enrolled patients included aortic aneurysm (abdominal, n = 8; thoracic, n = 9); aortic dissection (n = 20); aortic stenosis (n = 24), aortic regurgitation (n = 10), and mitral stenosis/regurgitation (n = 17). In total, 10 (9.7%) of the 103 specimens showed IgG4-positive cell infiltration with various intensities; five of these were aortic valve specimens from aortic stenosis, and IgG4-positive cell infiltration was present at >10 /HPF in three of them. In one aortic wall sample from an abdominal aortic aneurysm, various histopathological features of IgG4-RD, such as IgG4-positive cell infiltration, obliterating phlebitis, and storiform fibrosis, were observed.ConclusionsIgG4-positive cell infiltration was observed in 9.7% of the surgical cardiovascular specimens, mainly in the aortic valve from aortic stenosis and in the aortic wall from aortic aneurysm. Whether IgG4-positive cell infiltration has pathophysiological importance in the development or progression of cardiovascular diseases should be investigated in future studies.

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Section: Discussionmentioning

confidence: 99%

IgG4-positive cell infiltration in various cardiovascular disorders - results from histopathological analysis of surgical samples

Hourai

Kasashima

Sohmiya

et al. 2017

BMC Cardiovasc Disord

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“…The chronic lymphadenitis and lymphoid hyperplasia associated with the disease are mostly detected in lymph node biopsies, so there are some deficiencies in confirming CD via needle biopsy. Based on the diagnostic criteria for CD, with the exception of the characteristic clinicopathological changes of patients, other related primary changes are often excluded in clinical diagnosis, thus, connective tissue disease, tumor disease, and infectious disease should be excluded in clinical diagnosis of CD, so as to improve the accuracy, sensitivity and specificity of clinical diagnosis [ 20 ]. The limitations in this study were that the potential value of IL-6 and CRP as diagnostic markers of CD children requires further validation within a large sample size, and by using comparison of histological and immunohistochemical analysis, including staining for CD3, CD5, CD10, CD20, CD23, CD79, and Ki-67 as previously reported [ 21 ].…”

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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“…Two years after rituximab treatment, the patient's disease was stable with PSL (0.2 mg/kg). Izumi et al [22] reported a 50-year-old woman with characteristic feat u r e s o f M C D , i n c l u d i n g p o l y c l o n a l hypergammaglobulinemia, multiple enlarged lymph nodes, and pulmonary interstitial infiltration, with elevated serum IgG4 level and a 37.3% IgG4+/IgG+ plasma cell ratio in her biopsy specimen, while without organ involvement of IgG4-RD, corticosteroid treatment resolved the serological and imaging abnormalities. The 14 IgG4-CD patients in present study showed relatively favorable response to glucocorticoid and immunosuppressant therapy, one untreated IgG4-CD patient was stable, during a 33 (4-56)-month follow-up, and this was the same as those reported in the literature.…”

Section: Discussionmentioning

confidence: 99%

Clinical characteristics of a concurrent condition of IgG4-RD and Castleman’s disease

et al. 2018

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IgG4-related disease (IgG4-RD) and Castleman's disease (CD) share similar clinical manifestations. When the histopathology coincides with the diagnosis of both IgG4-RD and CD, it is hard to depart the two disease entities utterly; here we call it IgG4-CD provisionally. In this study, we aim to review the clinical features of IgG4-CD. This study is based on a retrospective analysis of a prospectively acquired database. IgG4-CD was defined histopathologically in patients who fulfilled the diagnosis of both IgG4-RD and CD. Forty-five definite IgG4-RD and 16 multicentric CD (MCD) patients were recruited as controls. Clinical features including organ involvement, serum IgG4, IgG, IgE, ESR, CRP, and IL-6 levels were collected and analyzed. Fifteen patients (2.8%) out of 534 patients with IgG4-RD in China's largest prospective IgG4-RD and Mimicry cohort fulfilled the definition of IgG4-CD. There were 14 males and 1 female, whose mean age was 47 ± 18 years old, and the median disease duration before diagnosis was 12 (1-132) months. Eight patients have allergic disease history. IgG4-CD patients had more lymph node involvement (100 vs 57.8%, P < 0.01), while IgG4-RD patients had more submandibular (33.3 vs 77.1%, P < 0.01) and parotid gland (13.3 vs 40.9%, P < 0.05) affected. IgG4-CD patients had significantly higher levels of ESR, CRP, IgG, IgG1, IgG3, IgG4, and IgE than IgG4-RD patients. Compared with MCD patients, IgG4-CD patients showed higher incidence of salivary gland and paranasal sinus involvement, higher hemoglobin, eosinophil count, serum IgG4 level and IgG4/IgG ratio, and lower CRP, IL-6 levels, and IgG1/IgG, IgG2/IgG ratio. All patients with IgG4-CD exhibited relatively favorable outcomes. Both IgG4-RD and CD can involve multiple organs. There are a small group of patients who had clinical and pathological characteristics of both CD and IgG4-RD showed better clinical outcome. In the long-term prognosis of these patients, the relationship of CD and IgG4-RD is waiting to be further elucidated.

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Multicentric Castleman disease mimicking IgG4-related disease: A case report

Cited by 11 publications

References 20 publications

IgG4-positive cell infiltration in various cardiovascular disorders - results from histopathological analysis of surgical samples

IgG4-positive cell infiltration in various cardiovascular disorders - results from histopathological analysis of surgical samples

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

Clinical characteristics of a concurrent condition of IgG4-RD and Castleman’s disease

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