Successful treatment of refractory TAFRO syndrome with elevated vascular endothelial growth factor using thyroxine supplements

Oka, Shiro; Ôno, Kazuo; Nohgawa, Masaharu

doi:10.1002/ccr3.1430

Cited by 9 publications

(8 citation statements)

References 22 publications

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“…This study extends our previous study of TAFRO syndrome (7). To our knowledge, this is the first study that demonstrates the relationship between subclinical hypothyroidism and TAFRO syndrome with VEGF elevation.…”

Section: Discussionsupporting

confidence: 90%

“…Based on the overlap between iMCD and autoimmune diseases, autoimmunity may be responsible for initiating or perpetuating the cytokine storm in iMCD via an autoantibody antigenic stimulation. There is only one report describing the clinical significance of hypothyroidism in TAFRO syndrome (7), and subclinical hypothyroidism with the presence of autoantibodies suggests that autoimmunity is a pathological cause of TAFRO syndrome.…”

Section: Discussionmentioning

confidence: 99%

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Subclinical Hypothyroidism in TAFRO Syndrome

2019

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Objective TAFRO syndrome is rare, and its underlying mechanisms currently remain unknown. Furthermore, standard therapeutic strategies have yet to be established. One of the hallmarks of TAFRO is pathological hypercytokinemia, which involves vascular endothelial growth factor (VEGF). A correlation has been reported between elevated VEGF and TSH levels in patients with hypothyroidism. Although hypothyroidism is a common endocrine abnormality, its clinical significance in TAFRO syndrome remains unclear. Methods and Patients We investigated six patients diagnosed with TAFRO syndrome and examined their thyroid function in detail to obtain a deeper understanding of its relationship with cytokines and the manifestations of thyroid abnormalities as well as their clinical significance in TAFRO syndrome. Results Five patients had subclinical hypothyroidism, while one had clinical hypothyroidism. Plasma VEGF levels were elevated in all patients, with a mean level of 256 pg/mL. Treatment with thyroxine supplements and immunotherapy or chemotherapy improved the symptoms of TAFRO syndrome without recurrence as well as increased the VEGF levels in three patients. Conclusion The present results suggest that subclinical hypothyroidism may be a potential factor in the pathogenesis and symptomatology of TAFRO syndrome with VEGF elevation.

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Section: Discussionsupporting

confidence: 90%

Section: Discussionmentioning

confidence: 99%

Subclinical Hypothyroidism in TAFRO Syndrome

2019

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“…A 12-year-old Japanese male was referred to us with pitting edema of lower limbs and high fever refractory to antibiotics for Some TAFRO syndrome cases refractory to tocilizumab have been reported, [6][7][8][9] but the timing of evaluating the efficacy of tocilizumab was too early. In randomized double-blind placebo-controlled trials of siltuximab for iMCD, a median time of 33 days was required to show a response to siltuximab in responders.…”

Section: A Pediatric Case Of Tocilizumab-resistant Tafro Syndrome Trementioning

confidence: 99%

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

Minamikawa

Matsumura

Sato

et al. 2020

Pediatric Blood & Cancer

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“…In addition, this patient had subclinical hypothyroidism and sexual hormone disorders during chemotherapy. It has been reported previously that patients with TAFRO syndrome may be associated with endocrine system disorders 18,19 . Due to scarceness of the disease, more studies and cases are needed to confirm the possible link between TAFRO syndrome and endocrine system disorders.…”

Section: Discussionmentioning

confidence: 96%

TAFRO syndrome with fatigue and abdominal distension as the first symptom: A case report

Zhou

Tang

Chen³

et al. 2022

Hematological Oncology

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TAFRO syndrome is a peculiar and rare type of multi‐centric Castleman's disease which contained a series of symptoms such as thrombocytopenia (T), anasarca (A), fever (F), reticulin fibrosis (R), and organomegaly (O). Here we provide a case of TAFRO syndrome with the manifestation of fatigue, abdominal distension, and low fever at primary diagnosis, characterized by multiple lymphadenopathy of superficial mediastinal and retroperitoneal lymph nodes, and it was finally confirmed by lymph node biopsy. The patient recovered speedy after receiving CHOP chemotherapy. In this case report, the patient has a history of dust‐exposure and hepatitis B virus infection, which may be potentially related to the disease. In addition, this case suggests the importance of pathological biopsy of complete lymph nodes in diagnostic process.

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Successful treatment of refractory TAFRO syndrome with elevated vascular endothelial growth factor using thyroxine supplements

Cited by 9 publications

References 22 publications

Subclinical Hypothyroidism in TAFRO Syndrome

Subclinical Hypothyroidism in TAFRO Syndrome

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

TAFRO syndrome with fatigue and abdominal distension as the first symptom: A case report

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