Multicentric Castleman Disease Presenting with Fever

Smith, Christiana; Lee‐Miller, Cathy; Dishop, Megan K.; Cost, Carrye R.; Wang, Michael; Asturias, Edwin J.

doi:10.1016/j.jpeds.2014.08.042

Cited by 9 publications

(5 citation statements)

References 20 publications

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“…One study found that tocilizumab was efficacious in two children with multicentric Castleman’s disease who developed sustained fever after treatment with an IL-1 agonist or IVIG [65]. A 21-month-old boy with multicentric Castleman’s disease responded incompletely to initial tocilizumab treatment, but improved after subsequent chemotherapy [66]. In another case, a 16-year-old male was diagnosed with multicentric Castleman’s disease associated with abdominal pain, fatigue, weakness, fever, and night sweats [67].…”

Section: Resultsmentioning

confidence: 99%

Off-label use of tocilizumab to treat non-juvenile idiopathic arthritis in pediatric rheumatic patients: a literature review

et al. 2018

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Tocilizumab, an anti-interleukin-6 (IL-6) agent, is indicated as a treatment for several autoimmune or inflammatory diseases, including rheumatoid arthritis and juvenile idiopathic arthritis (JIA). IL-6 plays roles in both immune system dysregulation and inflammation, and thus efforts to extend the utility of tocilizumab in patients with autoinflammatory conditions are ongoing. Here, we survey the literature on the off-label use of tocilizumab in patients with juvenile-onset rheumatic diseases including juvenile systemic lupus erythematosus (SLE), juvenile dermatomyositis (DM), vasculitis, juvenile scleroderma, and other autoinflammatory diseases. There is no real evidence that tocilizumab is useful for patients with SLE and juvenile DM, but several cases of childhood Takayasu arteritis have experienced promising outcomes. In juvenile-onset scleroderma, for which no therapy that can halt disease progression is available, tocilizumab may stop progression and the associated functional impairment. Tocilizumab prevents systemic inflammation in patients with Kawasaki’s disease, but may develop coronary aneurysms. Tocilizumab has been used to treat several pediatric autoinflammatory diseases, including JIA-associated uveitis and Castleman’s disease. Further work in larger populations is necessary to confirm the effects of tocilizumab in patients with pediatric rheumatic diseases.

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Section: Resultsmentioning

confidence: 99%

Off-label use of tocilizumab to treat non-juvenile idiopathic arthritis in pediatric rheumatic patients: a literature review

et al. 2018

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“…Notably, 14/18 (78%) iMCD‐TAFRO patients responded to a combination including anti‐IL‐6 therapy (siltuximab or tocilizumab) and 9/13 (69%) responded to anti‐IL‐6 therapy±corticosteroids, according to the case report authors' assessments; three patients who responded to anti‐IL‐6 ± corticosteroids relapsed before the time of publication (Table 3). 17‐29 We also assessed all of the iMCD‐TAFRO and iMCD‐NOS cases with HyperV or HV histopathology at UAMS that were treated with anti‐IL‐6 therapy±corticosteroids. Here, 4/4 iMCD‐TAFRO and 7/7 iMCD‐NOS patients responded, according to the investigator's assessment of clinical and laboratory abnormalities.…”

Section: Resultsmentioning

confidence: 99%

“…therapy (siltuximab or tocilizumab) and 9/13 (69%) responded to anti-IL-6 therapy±corticosteroids, according to the case report authors' assessments; three patients who responded to anti-IL-6 ± corticosteroids relapsed before the time of publication (Table 3). [17][18][19][20][21][22][23][24][25][26][27][28][29] We also assessed all of the iMCD-TAFRO and iMCD- Anti-IL-6 + rituximab or cyclosporine ± CS iMCD-TAFRO 5 5 3…”

Section: Real-world Datamentioning

confidence: 99%

Insufficient evidence exists to use histopathologic subtype to guide treatment of idiopathic multicentric Castleman disease

Fajgenbaum

Goodman

et al. 2020

American J Hematol

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“…1,18 The majority of CD cases are adults, particularly females, with less than 100 cases reported in children (especially amongst teenage girls). 1,18,20,21 These adults affected are found within the third to fourth decade for UCD and fourth to fifth decade for MCD, with UCD accounting for approximately 87% of CD cases. 1,18 The hyaline vascular (HV) subtype of CD is characterized histomorphologically by prominent vascular proliferation and hyalinization of vessel walls admixed with variable follicular patterns such as lollipop follicles, onion skin mantle zone, and mantle zones fusion with twinning of germinal centers.…”

Section: Discussionmentioning

confidence: 99%

Multicentric Plasma-Cell Type Castleman Disease Masquerading As Hodgkin Lymphoma: A Case Report

Ekwere

Eziagu

2020

J Lab Physicians

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Castleman disease (CD), or angiofollicular hyperplasia, or giant lymph node hyperplasia, is a heterogeneous benign lymphoproliferative disorder of unknown etiology. It has three distinct histologic subtypes (hyaline vascular, plasma cell, and mixed hyaline vascular plasma cell types) as well as unicentric Castleman disease (UCD) and multicentric Castleman disease (MCD) variants. In the unicentric form, the disease is confined to one anatomical lymph node and usually with no systemic symptoms. However, in the multicentric form (further subdivided into idiopathic MCD, human herpes virus-8-associated MCD, and POEMS-associated MCD), lymphadenopathy is more generalized with more aggressive systemic symptoms mimicking a malignant lymphoma. Therefore, this case report aims to underscore the importance of immunohistochemical evaluation as an indispensable ancillary technique to routine histopathological examination of a lymph node biopsy specimen, as a gold standard for definitive diagnosis of proliferative lymph node lesions.

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Multicentric Castleman Disease Presenting with Fever

Cited by 9 publications

References 20 publications

Off-label use of tocilizumab to treat non-juvenile idiopathic arthritis in pediatric rheumatic patients: a literature review

Off-label use of tocilizumab to treat non-juvenile idiopathic arthritis in pediatric rheumatic patients: a literature review

Insufficient evidence exists to use histopathologic subtype to guide treatment of idiopathic multicentric Castleman disease

Multicentric Plasma-Cell Type Castleman Disease Masquerading As Hodgkin Lymphoma: A Case Report

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