Phase I/II Trial of Liver-derived Mesenchymal Stem Cells in Pediatric Liver-based Metabolic Disorders: A Prospective, Open Label, Multicenter, Partially Randomized, Safety Study of One Cycle of Heterologous Human Adult Liver-derived Progenitor Cells (HepaStem) in Urea Cycle Disorders and Crigler-Najjar Syndrome Patients

Transplantation. 2019 Sep;103(9):1903-1915. doi: 10.1097/TP.0000000000002605.

Abstract

Background: Regenerative medicine using stem cell technology is an emerging field that is currently tested for inborn and acquired liver diseases.

Objective: This phase I/II prospective, open label, multicenter, randomized trial aimed primarily at evaluating the safety of Heterologous Human Adult Liver-derived Progenitor Cells (HepaStem) in pediatric patients with urea cycle disorders (UCDs) or Crigler-Najjar (CN) syndrome 6 months posttransplantation. The secondary objective included the assessment of safety up to 12 months postinfusion and of preliminary efficacy.

Methods: Fourteen patients with UCDs and 6 with CN syndrome were divided into 3 cohorts by body weight and intraportally infused with 3 doses of HepaStem. Clinical status, portal vein hemodynamics, morphology of the liver, de novo detection of circulating anti-human leukocyte antigen antibodies, and clinically significant adverse events (AEs) and serious adverse events to infusion were evaluated by using an intent-to-treat analysis.

Results: The overall safety of HepaStem was confirmed. For the entire study period, patient-month incidence rate was 1.76 for the AEs and 0.21 for the serious adverse events, of which 38% occurred within 1 month postinfusion. There was a trend of higher events in UCD as compared with CN patients. Segmental left portal vein thrombosis occurred in 1 patient and intraluminal local transient thrombus in a second patient. The other AEs were in line with expectations for catheter placement, cell infusion, concomitant medications, age, and underlying diseases.

Conclusions: This study led to European clinical trial authorization for a phase II study in a homogeneous patient cohort, with repeated infusions and intermediate doses.

Publication types

  • Clinical Trial, Phase I
  • Clinical Trial, Phase II
  • Multicenter Study
  • Randomized Controlled Trial
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Age Factors
  • Child
  • Child, Preschool
  • Crigler-Najjar Syndrome / blood
  • Crigler-Najjar Syndrome / diagnosis
  • Crigler-Najjar Syndrome / drug therapy*
  • Crigler-Najjar Syndrome / physiopathology
  • Europe
  • Female
  • Humans
  • Infant
  • Liver / metabolism*
  • Liver / pathology
  • Liver / physiopathology
  • Liver Regeneration
  • Liver Transplantation* / adverse effects
  • Male
  • Prospective Studies
  • Stem Cell Transplantation* / adverse effects
  • Time Factors
  • Transplantation, Heterologous
  • Treatment Outcome
  • Urea Cycle Disorders, Inborn / blood
  • Urea Cycle Disorders, Inborn / diagnosis
  • Urea Cycle Disorders, Inborn / physiopathology
  • Urea Cycle Disorders, Inborn / surgery*

Associated data

  • EudraCT/2011-004074-28