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2018 ; 7
(1
): 162-168
Nephropedia Template TP
gab.com Text
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English Wikipedia
TAFRO syndrome with refractory thrombocytopenia responding to tocilizumab and
romiplostim: a case report
#MMPMID29468524
Noda-Narita S
; Sumida K
; Sekine A
; Hoshino J
; Mise K
; Suwabe T
; Hayami N
; Yamanouchi M
; Ueno T
; Mizuno H
; Kawada M
; Hiramatsu R
; Hasegawa E
; Sawa N
; Takaichi K
; Ohashi K
; Fujii T
; Ubara Y
CEN Case Rep
2018[May]; 7
(1
): 162-168
PMID29468524
show ga
Thrombocytopenia, anasarca, fever, reticulin fibrosis, organomegaly (TAFRO)
syndrome is a unique clinicopathologic subtype of multicentric Castleman's
disease that has recently been identified in Japan. However, little is known
about its renal histological changes and the optimal treatment for TAFRO
syndrome. An 80-year-old Japanese woman was admitted to our hospital for
evaluation of severe anasarca and weight gain (10 kg in a month). She had
polyneuropathy, monoclonal plasma cell proliferative disorder with positive kappa
M-protein, a sclerotic bone lesion, elevation of vascular endothelial growth
factor (VEGF), skin changes, and extravascular volume overload, which fulfilled
the diagnostic criteria for POEMS (polyneuropathy, organomegaly, endocrinopathy,
and monoclonal protein, skin changes) syndrome. However, kappa-type M-protein and
thrombocytopenia with positivity of platelet-associated immunoglobulin G antibody
were unusual, and fitted the diagnostic criteria for TAFRO syndrome. Renal biopsy
showed diffuse endocapillary proliferative glomerulonephritis with endothelial
swelling and the infiltration of monocytes and neutrophils without specific
immunoglobulin deposits. Her systemic symptoms were refractory to initial
treatment with high-dose melphalan and glucocorticoids. Alternative therapy with
an anti-interleukin-6 (IL-6) receptor antibody (tocilizumab) effectively
controlled the symptoms, while a thrombopoietin receptor agonist (romiplostim)
was effective for her thrombocytopenia. Results suggest that IL-6-VEGF axis and
an autoimmune mechanism may be responsible for TAFRO syndrome with clinical
features of POEMS and refractory thrombocytopenia, which can be successfully
treated with combination of tocilizumab and romiplostim.