Reversal of cardiomyopathy with tocilizumab in a case of <scp>HIV</scp>‐negative <scp>C</scp>astleman's disease

Man, Louise; Goudar, Ranjit K.

doi:10.1111/ejh.12161

Cited by 11 publications

(4 citation statements)

References 22 publications

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“…This result could imply a regional difference, but pulmonary involvement and secondary amyloidosis should also be carefully considered outside of Japan. Fourth, ECG abnormalities observed in 15.5% of patients in our study could be attributed to an underlying heart condition (Table 2); however concomitant heart conditions have been rarely reported [42]. There are reports of patients who developed CD-associated cardiac dysfunction as a result of heart amyloidosis and pulmonary arterial hypertension [43,44].…”

Section: Discussionmentioning

confidence: 56%

Clinicopathologic characteristics of 342 patients with multicentric Castleman disease in Japan

Murakami

Johkoh

Hayashi

et al. 2020

Modern Rheumatology

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Objectives: To assess the clinicopathologic features of Multicentric Castleman disease (MCD) patients in Japan. Methods: We assessed baseline data for 342 Japanese MCD patients with a biopsy-proven diagnosis, enrolled in a prospective, observational study for tocilizumab treatment. Results: Of 342 patients, 86.0% had plasma-cell type. None had a family history of MCD. Median disease duration of MCD was 3.7 years. Mean body weight and body mass index tended to be lower than those in the general Japanese population. The most common clinical presentations besides lymphadenopathy included fatigue (61.7%), pulmonary involvement (42.7%), and splenomegaly (41.8%). Secondary amyloidosis was reported in 34 patients (9.9%). Laboratory abnormalities included decreased hemoglobin and albumin, and increased acute-phase proteins, serum immunoglobulins, and interleukin-6 (IL-6). IL-6 levels among the MCD patients tested in this study were correlated with levels of albumin, hemoglobin, triglyceride, total cholesterol, C-reactive protein, fibrinogen and immunoglobulin G (Spearman's correlation coefficient, jrj ¼ 0.28-0.59). Conclusion: The clinical features and laboratory abnormalities are similar to those previously reported in other countries, besides higher rates of pulmonary involvement, secondary amyloidosis, and ECG abnormalities. Our results imply that IL-6 is involved in MCD pathogenesis. These findings would be informative for diagnosis and appropriate treatment for MCD. ARTICLE HISTORY

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Section: Discussionmentioning

confidence: 56%

Clinicopathologic characteristics of 342 patients with multicentric Castleman disease in Japan

Murakami

Johkoh

Hayashi

et al. 2020

Modern Rheumatology

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“…In some cases, cyclosporine A (CyA) is effective (see Table 2), which suggests Our case is the first report of TAFRO syndrome with cardiomyopathy. In cases of MCD, 7 cases with cardiac complications have been reported to date 17,18) . Diffuse hypokinesis of LV wall motion was evident on echocardiograms in all these cases.…”

Section: Discussionmentioning

confidence: 99%

Successful treatment by rituximab in a patient with TAFRO syndrome with cardiomyopathy

Hiramatsu¹,

Ohmura²,

Tsuji³

et al. 2016

Jpn. J. Clin. Immunol.

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TAFRO syndrome is a newly defined disease entity which is characterized by thrombocytopenia, anasarca, myelofibrosis, renal dysfunction, and organomegaly. A histological pattern of multiple lymphadenopathy of atypical Castleman's disease (CD) is also an important characteristic. A 48-year-old man was referred to our hospital with fever, asthenia, bilateral pleural effusion, ascites, generalized edema, dyspnea, hypoalbuminemia, severe thrombocytopenia, anemia, renal failure and proteinuria, whereas bacterial culture and serological and PCR tests for various viruses were all negative. A CT scan showed multiple lymphadenopathy and tissue sampling of inguinal lymph nodes showed a compatible histology with plasma cell type CD. A diagnosis of TAFRO syndrome was made. Ten days after hospitalization, sudden cardiac insufficiency and anuria developed. Despite glucocorticoid pulse therapy, tocilizumab and plasmapheresis, clinical and laboratory features did not improve. On the 34(th) hospital day, we started rituximab. His general condition started to improve in several days, and by one month later anasarca had improved drastically. Thrombocytopenia and renal function gradually improved and finally normalized. Cardiac motion also improved. This is the first report of a TAFRO syndrome patient with cardiomyopathy, who was successfully treated with rituximab.

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“…There are no reported cases of restrictive cardiomyopathy associated with CD, so this patient presented a unique challenge. There are few cases in the literature describing cardiomyopathy associated with CD, and all cases described are of dilated cardiomyopathies with ventricular systolic dysfunction, presumably due to severe systemic inflammation (3,(7)(8)(9). Slow improvement in dilated cardiomyopathy was reported in two cases with the use of the IL-6 receptor-blocking antibody tocilizumab (3,7).…”

Section: Discussionmentioning

confidence: 99%

“…There are few cases in the literature describing cardiomyopathy associated with CD, and all cases described are of dilated cardiomyopathies with ventricular systolic dysfunction, presumably due to severe systemic inflammation (3,(7)(8)(9). Slow improvement in dilated cardiomyopathy was reported in two cases with the use of the IL-6 receptor-blocking antibody tocilizumab (3,7). Without cardiac biopsy and with the dramatic FDG uptake on PET scan, we speculated the myocardium may be infiltrated by plasma cells with subsequent restrictive physiology.…”

Section: Discussionmentioning

confidence: 99%

The use of a Berlin Heart EXCOR LVAD in a child receiving chemotherapy for Castleman's disease

Thomas

Chandrakasan

O’Brien

et al. 2014

Pediatric Transplantation

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We present the unique case of a pediatric patient who received chemotherapy for a diagnosis of CD, while mechanically supported with a Berlin EXCOR LVAD secondary to restrictive cardiomyopathy. A four-yr-old previously healthy male with restrictive cardiomyopathy required MCS after cardiac arrest but was diagnosed with multicentric CD, a non-malignant lymphoproliferative disorder fueled by excessive IL-6 production. Treatment with IL-6 blockade (tocilizumab) every two wk and methylprednisolone had no effect on his lymph nodes or cardiac function while on temporary RotaFlow. A Berlin LVAD was placed for treatment with rituximab, COP, vincristine, and methylprednisolone. After three courses of chemotherapy, his inflammatory markers normalized and his lymphadenopathy decreased but cardiac function remained severely depressed. He tolerated chemotherapy on the Berlin but required frequent titrations of his anti-coagulation regimen and he did suffer a hemorrhagic stroke. His clinical status improved significantly with rehabilitation, and he tolerated heart transplantation without further complications. MCS is a feasible option as a bridge to recovery or heart transplant eligibility for patients with hemodynamic collapse requiring chemotherapy but it does necessitate close titration of the anti-coagulation regimen to coincide with changes in the inflammatory state.

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Reversal of cardiomyopathy with tocilizumab in a case of HIV‐negative Castleman's disease

Cited by 11 publications

References 22 publications

Clinicopathologic characteristics of 342 patients with multicentric Castleman disease in Japan

Clinicopathologic characteristics of 342 patients with multicentric Castleman disease in Japan

Successful treatment by rituximab in a patient with TAFRO syndrome with cardiomyopathy

The use of a Berlin Heart EXCOR LVAD in a child receiving chemotherapy for Castleman's disease

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