[Pseudo-adult Still's disease, anasarca, thrombotic thrombocytopenic purpura and dysautonomia: An atypical presentation of multicentric Castleman's disease. Discussion of TAFRO syndrome]

Rev Med Interne. 2016 Jan;37(1):53-7. doi: 10.1016/j.revmed.2015.04.006. Epub 2015 Sep 26.
[Article in French]

Abstract

Introduction: Multicentric Castleman's disease can mimic adult-onset Still disease. It is exceptionally associated with anasarca, thrombotic microangiopathy and dysautonomia.

Case report: We report a 32-year-old woman with an association of oligoanuria, anasarca, thrombotic microangiopathy with features compatible with adult-onset Still disease. The outcome was initially favorable with corticosteroids, immunoglobulins and plasmapheresis but with the persistence of relapses marked by severe autonomic syndrome and necessity of high dose corticosteroids. The diagnosis of mixed type Castleman's disease, HHV8 and HIV negative, was obtained four years after the onset of symptoms by a lymph node biopsy. The outcome was favorable after tocilizumab and corticosteroids but tocilizumab had to be switched to anakinra to ensure a proper and long-lasting control of the disease.

Conclusion: Our patient partially fits the description of TAFRO syndrome (Thrombocytopenia, Anasarca, myeloFibrosis, Renal dysfunction, Organomegaly), a MCM rare variant, recently described in Japanese patients.

Keywords: Adult-onset Still's disease; Dysautonomia; Dysautonomie; Maladie de Castleman multicentrique; Maladie de Still de l’adulte; Microangiopathie thrombotique; Multicentric Castleman's disease; Syndrome TAFRO; TAFRO syndrome; Thrombotic thrombocytopenic purpura.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Castleman Disease / complications
  • Castleman Disease / diagnostic imaging
  • Castleman Disease / pathology*
  • Diagnosis, Differential
  • Edema / diagnostic imaging
  • Edema / etiology
  • Edema / pathology*
  • Female
  • Humans
  • Primary Dysautonomias / diagnostic imaging
  • Primary Dysautonomias / etiology
  • Primary Dysautonomias / pathology*
  • Purpura, Thrombotic Thrombocytopenic / diagnostic imaging
  • Purpura, Thrombotic Thrombocytopenic / etiology
  • Purpura, Thrombotic Thrombocytopenic / pathology*
  • Radionuclide Imaging
  • Still's Disease, Adult-Onset / diagnostic imaging
  • Still's Disease, Adult-Onset / etiology
  • Still's Disease, Adult-Onset / pathology*
  • Syndrome

Supplementary concepts

  • Multi-centric Castleman's Disease