Castleman's disease with TAFRO syndrome: a case report from Syria

Alhoulaiby, Sami; Ahmad, Basel; Alrstom, Ali; Kudsi, Mayssoun

doi:10.1093/omcr/omx021

Cited by 7 publications

(6 citation statements)

References 22 publications

(31 reference statements)

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“…[ 1 ] Most cases of TAFRO syndrome have been reported in Japan, and more recently also in Europe, [ 3 , 4 ] North America, [ 5 , 6 ] South America, [ 7 ] and Western Asia. [ 8 ] As case reports have accumulated, its distinct clinical characteristics have become more obvious and in 2015, updated diagnostic criteria were proposed. [ 2 ] According to these criteria, the presence of all 3 major and at least 2 of the 4 minor criteria is necessary for diagnosis of TAFRO syndrome.…”

Section: Discussionmentioning

confidence: 99%

Clinicopathological features of progressive renal involvement in TAFRO syndrome

Tanaka

Tsujimoto²,

Yamamoto³

et al. 2017

Medicine

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Rationale:TAFRO syndrome is a systemic inflammatory disease characterized by a constellation of symptoms: Thrombocytopenia, Anasarca, MyeloFibrosis, Renal dysfunction, and Organomegaly. Progressive renal insufficiency is a predominant symptom; however, the mechanism of acute kidney injury (AKI) remains unclear, probably because severe thrombocytopenia prevents kidney biopsy. We report a rare case of TAFRO syndrome with histologically confirmed renal involvement.Patients concerns:A 70-year-old man developed fever, anasarca, AKI, thrombocytopenia, and hepatosplenomegaly.Diagnoses:Plasma vascular endothelial growth factor and serum interleukin-6 levels were significantly elevated. The diagnosis of TAFRO syndrome was made based on his clinical and laboratory findings. Kidney biopsy was performed for the evaluation of AKI and provided a diagnosis of membranoproliferative glomerulonephritis–like lesions due to endothelial injury. Glomerular capillary lumens were extremely narrowed or occluded by endothelial swelling, and marked widening of the subendothelial space by electron-lucent material resulted in mesangiolysis and a double-contoured glomerular basement membrane with no immune complex deposits.Interventions and Outcomes:The patient required temporary hemodialysis due to oliguric AKI, but steroid therapy rapidly improved renal function.Lessons:Typically, patients with progressive renal involvement in TAFRO syndrome rapidly develop oliguric or anuric AKI. This report suggests that the reduction of glomerular perfusion by glomerular endothelial injury might be a primary factor in the progressive AKI of TAFRO syndrome. Our case and the literature review indicate that steroid and/or biological therapies result in highly favorable renal outcomes in patients with progressive AKI in TAFRO syndrome.

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Section: Discussionmentioning

confidence: 99%

Clinicopathological features of progressive renal involvement in TAFRO syndrome

Tanaka

Tsujimoto²,

Yamamoto³

et al. 2017

Medicine

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“…TAFRO syndrome has been reported mostly in Japan, though it has recently been reported in other countries 7‐10 . TAFRO syndrome was first defined as a novel systemic inflammatory disease at a consensus meeting of medical professionals and researchers in Japan in 2012 1 .…”

Section: Discussionmentioning

confidence: 99%

“…TAFRO syndrome has been reported mostly in Japan, though it has recently been reported in other countries. [7][8][9][10] TAFRO syndrome was first defined as a novel systemic inflammatory disease at a consensus meeting of medical professionals and researchers in Japan in 2012. 1 Masaki et al proposed a classification for diagnostic criteria and disease severity based on 28 cases of TAFRO syndrome in 2015.…”

Section: F I G U R E 1 Chest Computed Tomography Shows Enlarged Lymph Nodes At the Bilateral Axillary Area (White Arrow Heads) (A) And Plmentioning

confidence: 99%

TAFRO syndrome presenting as intrahepatic cholangitis on autopsy

Nishioka

Nishino

Yoshizaki

et al. 2021

Clinical Case Reports

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“…TAFRO syndrome can occur in any age and any race, but it is more common in East Asian populations, especially Japanese and mostly occurs in the middle-aged and elderly (Alhoulaiby et al 2017;Coutier et al 2017;Finocchietto et al 2015;Kubokawa et al 2014;Liu et al 2016;Owattanapanich et al 2018). 7.3% (7/96) of HIVnegative CD and 15.9% (7/44) of MCD patients in our center were diagnosed with TAFRO syndrome, the rate was higher than the reported rate by Oksenhendler et al (2018) who described that 7% (2/27) of French iMCD consistent with TAFRO syndrome (Oksenhendler et al (2018), but lower than the results of Owattanapanich et al (2018) of 18.2% (6/33).…”

Section: Discussionmentioning

confidence: 99%

Clinical features and treatment of 7 Chinese TAFRO syndromes from 96 de novo Castleman diseases: a 10-year retrospective study

Zhang

Suo

Yang

et al. 2020

J Cancer Res Clin Oncol

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Background Castleman disease (CD) is a rare polyclonal lymphoproliferative disorder with unknown etiology. TAFRO syndrome is now regarded as a specific subtype of CD, and is still a huge challenge for clinicians. Methods To clarify the clinical features and management of TAFRO syndrome in China, we retrospectively analyzed 96 patients with HIV-negative CD (52 with unicentric CD and 44 with multicentric CD), who were diagnosed and treated at our center between 2008 and 2017. Specially, we systematically reviewed the 7 TAFRO syndrome cases based on the 2015 criteria proposed by Masaki. Results Among the 7 cases, there were 3 men and 4 women, and the median age was 53 years. The main symptoms included thrombocytopenia (7/7), anasarca (7/7), fever (4/7), renal dysfunction (7/7), and organomegaly (6/7). One patient was treated with corticosteroid monotherapy, one received RD (Rituximab, dexamethasone), and 5 received CHOP/COP like chemotherapy as first-line treatment, 2 of the 5 combined with Rituximab. Four patients needed hemodialysis or CRRT because of progressive renal failure. The outcome for TAFRO syndrome was significantly worse compared to other types of CD. Although 3 patients improved after early treatment, 4 patients died due to disease progression, and only one patient achieved complete resolution of all the symptoms after changing to lenalidomide based regimen. Conclusions This study reveals that TAFRO syndrome is more severe and has more systemic symptoms than other iMCD, most cases need active treatment, and their prognoses are poor. Lenalidomide based regimen may be as a promising new therapy for TAFRO syndrome.

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Castleman's disease with TAFRO syndrome: a case report from Syria

Cited by 7 publications

References 22 publications

Clinicopathological features of progressive renal involvement in TAFRO syndrome

Clinicopathological features of progressive renal involvement in TAFRO syndrome

TAFRO syndrome presenting as intrahepatic cholangitis on autopsy

Clinical features and treatment of 7 Chinese TAFRO syndromes from 96 de novo Castleman diseases: a 10-year retrospective study

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