The Outcome of Allogeneic Hematopoietic Stem Cell Transplantation for Inherited Diseases Is Influenced by HLA Match, Year of Transplantation, and Immunized Female Donor

Transplantation. 2019 Jun;103(6):1247-1252. doi: 10.1097/TP.0000000000002481.

Abstract

Background: For many inborn errors of metabolism (IEM), allogeneic hematopoietic stem cell transplantation (HSCT) is the only cure.

Methods: We report the outcome in 160 patients with inherited diseases, who were treated with HSCT in 3 decades. Median age was 3 years (range 0.1-63). Grafts were from matched related donors (MRDs, 56), matched unrelated donors (MUDs, 66), or HLA-mismatched donors (38).

Results: Graft failure (GF) occurred in 26 patients (16%), severe acute graft-versus-host disease (GVHD) in 9 (6%), and chronic GVHD in 23 (12%). Ten-year survival was 64% before the year 2000 and 86% after that (P = 0.01). Ten-year survival for MRD grafts was 90%, as opposed to 79% for MUD grafts and 56% for HLA-mismatched grafts (P = 0.03). In multivariate analysis, GF was associated with having an HLA-mismatched donor (P < 0.05) or MUD (P = 0.015) and with reduced-intensity conditioning (P < 0.01). Death was associated with year of transplant (P = 0.015), having an HLA-mismatched donor (P < 0.001), and being a male recipient from an immune female donor (P = 0.05).

Conclusions: The outcome after HSCT for IEM depends on HLA match, year and immune female donor.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Allografts
  • Child
  • Child, Preschool
  • Donor Selection*
  • Female
  • Genetic Diseases, Inborn / genetics
  • Genetic Diseases, Inborn / immunology
  • Genetic Diseases, Inborn / mortality
  • Genetic Diseases, Inborn / surgery*
  • Genetic Predisposition to Disease
  • Graft Survival
  • Graft vs Host Disease / immunology
  • Graft vs Host Disease / mortality
  • HLA Antigens / immunology*
  • Hematopoietic Stem Cell Transplantation* / adverse effects
  • Hematopoietic Stem Cell Transplantation* / mortality
  • Heredity
  • Histocompatibility*
  • Humans
  • Infant
  • Middle Aged
  • Risk Assessment
  • Risk Factors
  • Sex Factors
  • Time Factors
  • Treatment Outcome
  • Unrelated Donors
  • Young Adult

Substances

  • HLA Antigens