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Sjögren's syndrome manifesting as clinicopathological features of TAFRO syndrome: A case report.

Abstract TAFRO syndrome is a newly proposed disorder that manifests as thrombocytopenia, anasarca, fever, reticulin myelofibrosis, renal dysfunction, and organomegaly. In this report, we describe the development of severe TAFRO syndrome-like systemic symptoms during the clinical course of juvenile-onset Sjögren's syndrome in a 32-year-old woman.
PMID
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TAFRO Syndrome.

TAFRO syndrome with primary Sjogren's syndrome.

Authors

Mayor MeshTerms
Keywords
Journal Title medicine
Publication Year Start




PMID- 29390349
OWN - NLM
STAT- MEDLINE
DCOM- 20180212
LR  - 20180212
IS  - 1536-5964 (Electronic)
IS  - 0025-7974 (Linking)
VI  - 96
IP  - 50
DP  - 2017 Dec
TI  - Sjogren's syndrome manifesting as clinicopathological features of TAFRO syndrome:
      A case report.
PG  - e9220
LID - 10.1097/MD.0000000000009220 [doi]
AB  - RATIONALE: TAFRO syndrome is a newly proposed disorder that manifests as
      thrombocytopenia, anasarca, fever, reticulin myelofibrosis, renal dysfunction,
      and organomegaly. In this report, we describe the development of severe TAFRO
      syndrome-like systemic symptoms during the clinical course of juvenile-onset
      Sjogren's syndrome in a 32-year-old woman. PATIENT CONCERNS: The patient was
      admitted due to dyspnea, fever, polyarthralgia, and generalized edema. She had
      been diagnosed with Sjogren's syndrome at the age of 14 years, based on
      histopathological examination of a biopsy of the minor salivary glands and the
      development of Raynaud's phenomenon, with no follow-up treatment required. On
      admission, she presented with anemia, elevated C-reactive protein levels,
      anasarca, and hepato-splenomegaly. A bone marrow examination revealed increased
      megakaryocytes with reticulin fibrosis, and the histopathology of an axillary
      lymph node was consistent with mixed-type Castleman disease. Eventually, she
      developed thrombocytopenia. INTERVENTIONS: Her symptoms fulfilled all of the
      major and minor categories of the diagnostic criteria for TAFRO syndrome.
      However, considering her prior diagnosis, we assumed that the clinical
      presentation was consistent with an acute exacerbation of Sjogren's syndrome.
      Unlike typical cases of TAFRO syndrome, the administration of relatively low-dose
      prednisolone relieved her symptoms. LESSONS: Differentiation between TAFRO
      syndrome and exacerbation of an autoimmune disease is clinically important,
      although this can be challenging. Identification of specific biomarkers for TAFRO
      syndrome would be clinically beneficial.
CI  - Copyright (c) 2017 The Authors. Published by Wolters Kluwer Health, Inc. All
      rights reserved.
FAU - Fujimoto, Shino
AU  - Fujimoto S
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Kawabata, Hiroshi
AU  - Kawabata H
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Kurose, Nozomu
AU  - Kurose N
AD  - Department of Pathology and Laboratory Medicine, Kanazawa Medical University,
      Daigaku, Uchinada, Ishikawa-ken, Japan.
FAU - Kawanami-Iwao, Haruka
AU  - Kawanami-Iwao H
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Sakai, Tomoyuki
AU  - Sakai T
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Kawanami, Takafumi
AU  - Kawanami T
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Fujita, Yoshimasa
AU  - Fujita Y
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Fukushima, Toshihiro
AU  - Fukushima T
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
FAU - Masaki, Yasufumi
AU  - Masaki Y
AD  - Department of Hematology and Immunology, Kanazawa Medical University.
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Medicine (Baltimore)
JT  - Medicine
JID - 2985248R
RN  - 0 (Glucocorticoids)
RN  - 9PHQ9Y1OLM (Prednisolone)
RN  - Multi-centric Castleman's Disease
SB  - AIM
SB  - IM
MH  - Adult
MH  - Castleman Disease/*diagnosis/drug therapy
MH  - Diagnosis, Differential
MH  - Female
MH  - Glucocorticoids/therapeutic use
MH  - Humans
MH  - Prednisolone/therapeutic use
MH  - Sjogren's Syndrome/*diagnosis
EDAT- 2018/02/03 06:00
MHDA- 2018/02/13 06:00
CRDT- 2018/02/03 06:00
PHST- 2018/02/03 06:00 [entrez]
PHST- 2018/02/03 06:00 [pubmed]
PHST- 2018/02/13 06:00 [medline]
AID - 10.1097/MD.0000000000009220 [doi]
AID - 00005792-201712150-00099 [pii]
PST - ppublish
SO  - Medicine (Baltimore). 2017 Dec;96(50):e9220. doi: 10.1097/MD.0000000000009220.