A pediatric case of tocilizumab‐resistant TAFRO syndrome treated successfully with rituximab

Minamikawa, Shogo; Matsumura, Osamu; Sato, Seiko; Nakagishi, Yasuo; Tanaka, Ryojiro

doi:10.1002/pbc.28489

Cited by 7 publications

(12 citation statements)

References 26 publications

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“…Fortunately, our patient was able to go into remission with the use of tocilizumab and prednisone. However, there have been reports of TAFRO syndrome being difficult to control even with tocilizumab 3 . Interestingly, despite clear evidence of acute liver injury on CT and histological findings, our patient had no elevation in transaminase levels.…”

Section: Figurecontrasting

confidence: 54%

“…TAFRO syndrome is an idiopathic multicentric Castleman disease characterized by thrombocytopenia, anasarca, fever, reticulin fibrosis, and organomegaly on bone marrow biopsy. It was first described in 2010 by a Japanese hematologist; however, there are only a few reported pediatric cases 1–3 . We highlight a case of TAFRO syndrome, in which the imaging results were similar to those of acute hepatitis.…”

Section: Figurementioning

confidence: 75%

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TAFRO syndrome in an adolescent with acute hepatitis‐like imaging findings

Matsumoto,

Yamazaki,

Inage

et al. 2023

Pediatrics International

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Section: Figurecontrasting

confidence: 54%

Section: Figurementioning

confidence: 75%

TAFRO syndrome in an adolescent with acute hepatitis‐like imaging findings

Matsumoto,

Yamazaki,

Inage

et al. 2023

Pediatrics International

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“…iMCD‐TAFRO is an aggressive subtype of iMCD, typically affecting adults, 8 although it has been reported in pediatric cases 9,10 . IL‐6 is thought to be a primary driver of the systemic inflammation seen in iMCD‐TAFRO, 8 with VEGF also likely contributory to its development 11 .…”

Section: Discussionmentioning

confidence: 99%

Idiopathic multicentric Castleman disease with TAFRO clinical subtype responsive to IL‐6/JAK inhibition: A pediatric case series

Lust

Gong

Remiker

et al. 2021

Pediatric Blood & Cancer

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TAFRO (thrombocytopenia, anasarca, fever/elevated C‐reactive protein, reticulin myelofibrosis, renal dysfunction, and organomegaly) clinical subtype of idiopathic multicentric Castleman disease (iMCD‐TAFRO) is a rare lymphoproliferative disease characterized by systemic inflammation. First‐line treatment for iMCD‐TAFRO includes steroids and interleukin (IL)‐6 blockade. Many patients have refractory disease, which is associated with significant morbidity and mortality, and treatment remains challenging. We present two pediatric cases of iMCD‐TAFRO. One patient responded to IL‐6 blockade; the other was refractory to siltuximab and chemotherapy, ultimately responding to JAK inhibition with ruxolitinib. This is the first reported pediatric case of refractory iMCD‐TAFRO responding to JAK inhibition.

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“…Approximately two‐thirds of the patients achieved a response, showing that rituximab is an effective treatment for TAFRO syndrome patients in Western countries. The efficacy of rituximab has also been verified in pediatric patients 68 . Some case reports have shown that the use of the R‐CHOP regimen (rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisolone), 69,70 as used for invasive B‐cell lymphoma, can inhibit an overactivated immune system and prevent the occurrence of a cytokine storm.…”

Section: Treatment Managementmentioning

confidence: 99%

“…The efficacy of rituximab has also been verified in pediatric patients. 68 Some case reports have shown that the use of the R-CHOP regimen (rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisolone), 69,70 as used for invasive B-cell lymphoma, can inhibit an overactivated immune system and prevent the occurrence of a cytokine storm. In a retrospective study from China, five patients received CHOP/COP-like chemotherapy as the first-line treatment, among whom two patients also received rituximab; all patients eventually died due to disease progression and drug-related toxicity.…”

Section: Rituximabmentioning

confidence: 99%

TAFRO syndrome: A disease that known is half cured

Chen

Feng

Zhang

et al. 2022

Hematological Oncology

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Thrombocytopenia, anasarca, fever, reticulin fibrosis/renal failure, and organomegaly (TAFRO) syndrome is rare in clinical practice. It is a systemic inflammatory disease caused by a cytokine storm. Its clinical manifestations include thrombocytopenia, systemic edema, fever, bone marrow fibrosis, renal insufficiency, and organ enlargement. The high mortality rate of TAFRO syndrome is due to the difficulty of acquiring biopsy samples for diagnosis and the rapid disease progression. This disease is poorly understood by clinicians. Early detection, accurate diagnosis, and timely treatment play key roles in prolonging the survival of the patients. This review summarizes the latest progress in the pathogenesis, diagnostic criteria, and treatment regimens of TAFRO syndrome, aiming to help clinicians better understand TAFRO syndrome and improve its diagnosis and treatment.

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A pediatric case of tocilizumab‐resistant TAFRO syndrome treated successfully with rituximab

Cited by 7 publications

References 26 publications

TAFRO syndrome in an adolescent with acute hepatitis‐like imaging findings

TAFRO syndrome in an adolescent with acute hepatitis‐like imaging findings

Idiopathic multicentric Castleman disease with TAFRO clinical subtype responsive to IL‐6/JAK inhibition: A pediatric case series

TAFRO syndrome: A disease that known is half cured

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