Cord Blood Transplantation Following Reduced-intensity Conditioning for Adult-onset Inherited Hemophagocytic Lymphohistiocytosis

Intern Med. 2016;55(6):667-71. doi: 10.2169/internalmedicine.55.5241. Epub 2016 Mar 15.

Abstract

Inherited hemophagocytic lymphohistiocytosis (HLH) is a genetic anomaly disorder in which abnormally activated cytotoxic T lymphocytes cannot induce the apoptosis of target cells and antigen-presenting cells, leading to hemophagocytosis, pancytopenia, and a variety of symptoms such as a high fever. The present patient with adult-onset HLH developed refractory disease despite receiving immunosuppressive treatments. He underwent a reduced-intensity conditioning (RIC) regimen that comprised antithymocyte globulin (ATG) followed by cord blood transplantation (RIC-CBT). He achieved and maintained a complete donor type. The incorporation of ATG into RIC-CBT may prevent graft failure and control hemophagocytosis, however, further efforts are necessary to reduce infectious complications.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Age of Onset
  • Aged
  • Antilymphocyte Serum / therapeutic use*
  • Cord Blood Stem Cell Transplantation*
  • Epstein-Barr Virus Infections / complications
  • Epstein-Barr Virus Infections / therapy*
  • Epstein-Barr Virus Infections / virology
  • Fatal Outcome
  • Graft vs Host Disease / drug therapy*
  • Humans
  • Immunosuppressive Agents / therapeutic use*
  • Lymphohistiocytosis, Hemophagocytic / diagnosis
  • Lymphohistiocytosis, Hemophagocytic / genetics
  • Lymphohistiocytosis, Hemophagocytic / surgery*
  • Male
  • T-Lymphocytes, Cytotoxic
  • Transplantation Chimera
  • Transplantation Conditioning / methods

Substances

  • Antilymphocyte Serum
  • Immunosuppressive Agents