Transplantation for congenital heart disease is associated with an increased risk of Epstein-Barr virus-related post-transplant lymphoproliferative disorder in children

J Heart Lung Transplant. 2021 Jan;40(1):24-32. doi: 10.1016/j.healun.2020.10.006. Epub 2020 Oct 26.

Abstract

Background: Children undergoing heart transplant are at higher risk of developing post-transplant lymphoproliferative disorder (PTLD) than other solid organ recipients. The factors driving that risk are unclear. This study investigated risk factors for PTLD in children transplanted at 1 of 2 United Kingdom pediatric cardiac transplantation centers.

Methods: All children (<18 years, n = 200) transplanted at our institution over a 16-year period were analyzed. Freedom from PTLD was assessed using the Kaplan-Meier method and Cox proportional regression.

Results: PTLD occurred in 17 of 71 children transplanted for congenital heart disease (CHD) and 18 of 129 transplanted for acquired cardiomyopathy (ACM). The cumulative incidence of all PTLD was 21.1% at 5 years after transplant. Median time from transplant to PTLD was 2.9 years (interquartile range: 0.9-4.6). Negative Epstein-Barr virus (EBV) serostatus pre-transplant (adjusted hazard ratio [HR]: 2.7, 95% CI: 1.3-5.6, p = 0.01) and underlying CHD (adjusted HR: 3.2, 95% CI: 1.4-7.4, p = 0.007) were independently associated with higher risk of PTLD. Age at thymectomy was significantly different between children with CHD and ACM (0.4 vs 5.5 years, p < 0.01). Median CD4+ and CD8+ T lymphocyte counts at 2 years after transplant were significantly lower in children transplanted for CHD vs ACM (CD4+: 391/µl vs 644/µl, p = 0.01; CD8+: 382/µl vs 500/µl, p = 0.01). At 5 years after transplant, those differences persisted among patients who developed PTLD (CD4+, 430/µl vs 963/µl, p < 0.01 and CD8+, 367/µl vs 765/µl, p < 0.01).

Conclusion: Underlying CHD is an independent risk factor for PTLD and is associated with a younger age at thymectomy. A persistent association with altered T lymphocyte subsets may contribute to the impaired response to primary EBV infection and increase the risk of PTLD.

Publication types

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

MeSH terms

  • Child
  • Child, Preschool
  • Epstein-Barr Virus Infections / epidemiology
  • Epstein-Barr Virus Infections / etiology*
  • Epstein-Barr Virus Infections / virology
  • Female
  • Follow-Up Studies
  • Heart Defects, Congenital / surgery*
  • Heart Transplantation / adverse effects*
  • Herpesvirus 4, Human*
  • Humans
  • Incidence
  • Infant
  • Lymphoproliferative Disorders / epidemiology
  • Lymphoproliferative Disorders / etiology*
  • Lymphoproliferative Disorders / virology
  • Male
  • Postoperative Complications / epidemiology
  • Postoperative Complications / etiology*
  • Postoperative Complications / virology
  • Retrospective Studies
  • Risk Assessment / methods*
  • Risk Factors
  • United Kingdom / epidemiology