Haploidentical Hematopoietic Cell Transplantation with Post-Transplant Cyclophosphamide in a Patient with Chronic Granulomatous Disease and Active Infection: A First Report

J Clin Immunol. 2015 Oct;35(7):675-80. doi: 10.1007/s10875-015-0204-y. Epub 2015 Oct 9.

Abstract

Purpose: We describe haploidentical hematopoietic cell transplantation (HCT) with high-dose post-transplant cyclophosphamide (PTCy) in a boy with x-linked chronic granulomatous disease (CGD).

Methods: A persistent and life-threatening fungal infection was the indication for HSCT. Non-myeloablative conditioning with PTCy (50 mg/kg days 3 and 4) was used in the absence of fully matched donors.

Results: Engraftment occurred on day 24. The patient experienced Grade 2 graft-versus-host disease of the skin and gastrointestinal tract and CMV infection, both of which were controlled. Chimerism was 100 % at days 30 and 6 months. Cessation of antifungal therapy was consistent with cure of the infection.

Conclusions: Haploidentical HCT with high-dose PTCy for CGD is feasible and succeeded even in the context of active infection.

Keywords: Chronic granulomatous disease; graft versus host disease; haploidentical; hematopoietic cell transplantation; post-transplant cyclophosphamide; scedosporium apiospermum.

Publication types

  • Case Reports
  • Research Support, N.I.H., Extramural
  • Research Support, N.I.H., Intramural

MeSH terms

  • Blood Transfusion, Autologous*
  • Chimerism / drug effects
  • Cyclophosphamide / administration & dosage*
  • Drug Dosage Calculations
  • Graft vs Host Disease / etiology
  • Graft vs Host Disease / prevention & control*
  • Granulomatous Disease, Chronic / complications
  • Granulomatous Disease, Chronic / therapy*
  • HLA Antigens / immunology
  • Humans
  • Immunosuppressive Agents / administration & dosage*
  • Infant
  • Male
  • Mycoses / etiology
  • Mycoses / therapy*
  • National Institutes of Health (U.S.)
  • Postoperative Complications / prevention & control*
  • Scedosporium*
  • Transplantation Conditioning
  • United States

Substances

  • HLA Antigens
  • Immunosuppressive Agents
  • Cyclophosphamide