Near‐total resolution of multicentric Castleman disease by prolonged treatment with thalidomide

Starkey, Cindi R.; Joste, Nancy E.; Lee, Fa-Chyi

doi:10.1002/ajh.20538

Cited by 30 publications

(24 citation statements)

References 2 publications

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“…However, symptoms are found to recur soon when the antibody is discontinued. Some new-targeted agents, such as thalidomide [5,29,30] and bortezomib [31], are also in study for MCD therapy and may be used by more patients in the future.…”

Section: Discussionmentioning

confidence: 99%

Multicentric Castleman’s disease presenting with multiple nodes in lungs: a case report and literature review

Sun

Han

2008

Int J Hematol

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We report a rare multicentric Castleman's disease case whose lung manifestation is severe than any known report. This is a 64-year-old woman, she had repeated cough, fever and breathlessness for more than 1 year. CT imaging showed multiple small nodules in both the lungs at the first onset. Large nodes and masses in both the lungs evolved 1 year later. After three times of lung biopsies (one video-assisted thoracoscopic surgery and two CT-guided percutaneous transthoracic), she was finally diagnosed as multicentric Castleman's disease. She got surprising relief after combination chemotherapy in spite of her severe lung changes.

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Section: Discussionmentioning

confidence: 99%

Multicentric Castleman’s disease presenting with multiple nodes in lungs: a case report and literature review

Sun

Han

2008

Int J Hematol

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“…97 Thalidomide, an immunomodulator that inhibits TNF-a, IL-1, IL-6, IL-12, and VEGF and stimulates T cells potentially via cereblon inhibition, has demonstrated effectiveness at inducing remission, decreasing IL-6 levels, and lowering C-reactive protein in iMCD. 66,98,99 Anakinra, an IL-1 receptor antagonist, has also been reported to induce remission in a pediatric case of iMCD and in a patient who did not respond to anti-IL-6 therapy. 60,100 Further elucidation of iMCD pathogenesis is urgently needed to provide additional candidate targeted therapies.…”

Section: Treatmentmentioning

confidence: 99%

HHV-8-negative, idiopathic multicentric Castleman disease: novel insights into biology, pathogenesis, and therapy

Fajgenbaum

Rhee

Nabel

2014

Blood

272

265

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Multicentric Castleman's disease (MCD) describes a heterogeneous group of disorders involving proliferation of morphologically benign lymphocytes due to excessive proinflammatory hypercytokinemia, most notably of interleukin-6. Patients demonstrate intense episodes of systemic inflammatory symptoms, polyclonal lymphocyte and plasma cell proliferation, autoimmune manifestations, and organ system impairment. Human herpes virus-8 (HHV-8) drives the hypercytokinemia in all HIV-positive patients and some HIV-negative patients. There is also a group of HIV-negative and HHV-8-negative patients with unknown etiology and pathophysiology, which we propose referring to as idiopathic MCD (iMCD). Here, we synthesize what is known about iMCD pathogenesis, present a new subclassification system, and propose a model of iMCD pathogenesis. MCD should be subdivided into HHV-8-associated MCD and HHV-8-negative MCD or iMCD. The lymphocyte proliferation, histopathology, and systemic features in iMCD are secondary to hypercytokinemia, which can occur with several other diseases. We propose that 1 or more of the following 3 candidate processes may drive iMCD hypercytokinemia: systemic inflammatory disease mechanisms via autoantibodies or inflammatory gene mutations, paraneoplastic syndrome mechanisms via ectopic cytokine secretion, and/or a non-HHV-8 virus. Urgent priorities include elucidating the process driving iMCD hypercytokinemia, identifying the hypercytokine-secreting cell, developing consensus criteria for diagnosis, and building a patient registry to track cases.

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“…Furthermore the proinflammatory factors IL1, IL6 and IL12 and Cox 2 are blocked and levels of antiinflammatory cytokines, including IL10 [20,23,24] are increased. These properties can be utilized in the treatment of leprosy [25], Morbus Castleman [26], a lymphoproliferative disease mainly driven by IL6 hypersecretion, in autoimmune diseases, including rheumatoid arthritis [27], lupus erythematodes [28] and Behcet's disease [29].…”

Section: Anti-inflammationmentioning

confidence: 99%

Cereblon binding molecules in multiple myeloma

Kortüm¹,

Zhu²,

Shi³

et al. 2015

Blood Reviews

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Near‐total resolution of multicentric Castleman disease by prolonged treatment with thalidomide

Cited by 30 publications

References 2 publications

Multicentric Castleman’s disease presenting with multiple nodes in lungs: a case report and literature review

Multicentric Castleman’s disease presenting with multiple nodes in lungs: a case report and literature review

HHV-8-negative, idiopathic multicentric Castleman disease: novel insights into biology, pathogenesis, and therapy

Cereblon binding molecules in multiple myeloma

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