Reversible cardiomyopathy associated with Multicentric Castleman disease: successful treatment with tocilizumab, an anti-interleukin 6 receptor antibody - PubMed (original) (raw)

Case Reports

Reversible cardiomyopathy associated with Multicentric Castleman disease: successful treatment with tocilizumab, an anti-interleukin 6 receptor antibody

Junya Kanda et al. Int J Hematol. 2007 Apr.

Abstract

Multicentric Castleman disease (MCD) is a rare lymphoproliferative disorder characterized by systemic lymphadenopathy and inflammatory symptoms that are associated with the overproduction of interleukin 6 (IL-6). Although several nonlymphoid organs can also be damaged in MCD, only a few cases with cardiac complications have been reported to date. We report a case of congestive heart failure in a female patient with MCD. On admission, her echocardiogram revealed a dilated and diffusely hypokinetic left ventricle. No stenosis was evident in the coronary angiogram. A histopathologic examination of a myocardial biopsy specimen showed mildly hypertrophic myocytes without infiltration of plasma cells or amyloid deposits. Repeated administration of an anti-IL-6 receptor antibody, tocilizumab (formerly known as MRA), gradually improved the ventricular wall motion over 6 months without any additional treatment for heart failure, suggesting the involvement of IL-6 in the pathogenesis of her cardiomyopathy. This report is the first of MCD complicated by heart failure treated successfully with tocilizumab. Administering tocilizumab in cases of MCD with unexplained cardiac dysfunction is worthwhile, because such a complication could be reversible.

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References

1. 1. Science. 1992 Jul 17;257(5068):387-9 - PubMed
2. 1. Blood. 2003 Apr 1;101(7):2496-506 - PubMed
3. 1. Heart Vessels. 2004 Sep;19(5):237-41 - PubMed
4. 1. Jpn Heart J. 2004 Mar;45(2):183-93 - PubMed
5. 1. J Biol Chem. 2003 May 2;278(18):16304-9 - PubMed

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