Multicentric Castleman's disease with reversible left ventricular diffuse hypokinesis.

Yamamoto, Satoshi; Nakatani, S; Kijima, Kazuhisa; Kohno, Keiko; Morita, Satoshi; Koito, Hitoshi; Yutaka, Hiroshi; Miyasaka, Youko; Nakamura, Shinya; Iwasaka, Toshiji; Uemura, Yoshiko

doi:10.2169/naika.90.123

Cited by 5 publications

(2 citation statements)

References 6 publications

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“…Van Rhee et al also reported that a rituximab treatment was relatively effective for iMCD-TAFRO [ 19 ]. Moreover, individual cases with iMCD or TAFRO syndrome with cardiomyopathy have been reported with the following treatments to improve cardiomyopathy: tocilizumab, tacrolimus, rituximab, steroids, CHOP-like chemotherapy, and rituximab plus CHOP-like chemotherapy [ 13 , 14 , 15 , 20 , 21 ]. Therefore, rituximab is an effective treatment option for TAFRO-like syndromes such as the present case.…”

Section: Discussionmentioning

confidence: 99%

Epstein–Barr Virus-Positive Cutaneous and Systemic Plasmacytosis with TAFRO Syndrome-like Symptoms Successfully Treated with Rituximab

et al. 2023

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Histopathologic findings in the lymph nodes of patients with thrombocytopenia, anasarca, fever, reticulin fibrosis, renal dysfunction, and organomegaly (TAFRO) syndrome are similar to those of idiopathic multicentric Castleman’s disease (iMCD), but TAFRO syndrome is different from iMCD in how it can progress rapidly and be fatal. These patients present scarce lymphadenopathy and low immunoglobulin levels. We present a case of cutaneous and systemic plasmacytosis (C/SP) that caused TAFRO syndrome-like symptoms which were successfully treated with rituximab. A 67-year-old woman presented with fever and a pruritic skin rash. Numerous plasma cells were observed in the peripheral blood and imaging revealed organomegaly, anasarca, and generalized lymphadenopathy. Subsequently, she rapidly developed thrombocytopenia as well as renal and heart failure. She tested positive for the Epstein–Barr virus (EBV), elevated immunoglobulins, and C/SP, which are also atypical for TAFRO syndrome, thereby complicating the diagnosis. However, after using the Japanese TAFRO Syndrome Research Group diagnostic criteria, we promptly administered rituximab to treat the C/SP with TAFRO-like symptoms and saved her life. Finally, histopathological observations of the lymph node biopsy helped confirm EBV-positive hypervascular-type iMCD. Therefore, diagnosing TAFRO-like syndromes based on the Japanese diagnostic criteria and following the associated treatment even without a confirmed diagnosis is crucial to improving the patient outcomes.

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Section: Discussionmentioning

confidence: 99%

Epstein–Barr Virus-Positive Cutaneous and Systemic Plasmacytosis with TAFRO Syndrome-like Symptoms Successfully Treated with Rituximab

et al. 2023

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“…Only a handful of cases of CD-associated cardiomyopathy have been reported worldwide (1,(9)(10)(11)(12)(13). Given the rarity of this disease and its cardiac complication, randomized therapeutic trials are not feasible.…”

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confidence: 99%

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

Man

Goudar

2013

European J of Haematology

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The association between Castleman's disease (CD) and cardiomyopathy has been rarely reported and the optimal therapeutic approach remains unknown. We report a previously healthy 20-year-old African American female who presented with fever, dyspnea, anasarca, and generalized lymphadenopathy. Diagnostic workup, including an axillary lymph node biopsy, revealed that she had human immunodeficiency virus-negative and human herpes virus-8-negative multicentric CD. She had a non-anaphylactoid infusion reaction during her fourth rituximab infusion. A few weeks later, she developed new-onset severe cardiomyopathy requiring inotropic therapy, warranting consideration for left ventricular assist device. Several clinical clues indicated her new-onset heart failure was a manifestation of her CD. Interestingly, a trial of tocilizumab (an anti-interleukin-6 receptor monoclonal antibody) resulted in complete resolution of her cardiomyopathy and other manifestations of CD. Tocilizumab received orphan drug approval for the treatment of CD in Japan, but is not yet approved for this indication in the United States. Clinicians should be aware of its potential clinical utility in CD.

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Aggressive TAFRO syndrome with reversible cardiomyopathy successfully treated with combination chemotherapy

et al. 2016

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TAFRO (thrombocytopenia, anasarca, myelofibrosis, renal dysfunction, and organomegaly) syndrome is an atypical manifestation of Castleman's disease. However, the mechanism underlying this very rare syndrome remains unknown, and there is no established standard treatment. Here we report cases of two young females with TAFRO syndrome who showed similar clinical courses. Both cases showed severe anasarca, ascites, and thrombocytopenia. Although high-dose steroids were ineffective, combination chemotherapy showed remarkable effects. However, both patients developed severe but reversible heart failure after CHOP therapy owing to diffuse cardiomyopathy, which was presumably associated with TAFRO syndrome. Therefore, although combination chemotherapy may be very effective in the treatment of TAFRO syndrome, careful observation for cardiomyopathy development is needed, particularly when using adriamycin-containing regimens.

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Multicentric Castleman's disease with reversible left ventricular diffuse hypokinesis.

Cited by 5 publications

References 6 publications

Epstein–Barr Virus-Positive Cutaneous and Systemic Plasmacytosis with TAFRO Syndrome-like Symptoms Successfully Treated with Rituximab

Epstein–Barr Virus-Positive Cutaneous and Systemic Plasmacytosis with TAFRO Syndrome-like Symptoms Successfully Treated with Rituximab

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

Aggressive TAFRO syndrome with reversible cardiomyopathy successfully treated with combination chemotherapy

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