Literature DB >> 31715057

Liver Transplantation in Children With Propionic Acidemia: Medium-Term Outcomes.

Richard Curnock1, Nigel D Heaton2, Hector Vilca-Melendez2, Anil Dhawan3, Nedim Hadzic3, Roshni Vara1,3.   

Abstract

Liver transplantation (LT) for patients with propionic acidemia (PA) is an emerging therapeutic option. We present a retrospective review of patients with PA who underwent LT at a tertiary liver center between 1995 and 2015. A total of 14 children were identified (8 males) with median age at initial presentation of 3 days (range, 0-77 days). Pretransplant median protein restriction was 1 g/kg/day (range, 0.63-1.75 g/kg/day), 71% required supportive feeding, and 86% had developmental delay. Frequent metabolic decompensations (MDs) were the main indication for LT with a median age at transplantation of 2.4 years (range, 0.8-7.1 years). Only 1 graft was from a living donor, and 13 were from deceased donors (4 auxiliary). The 2-year patient survival was 86%, and overall study and graft survival was 79% and 69%, respectively. Three patients died after LT: at 43 days (biliary peritonitis), 225 days (acute-on-chronic rejection with multiorgan failure), and 13.5 years (posttransplant lymphoproliferative disease). Plasma glycine and propionylcarnitine remained elevated but reduced after transplant. Of 11 survivors, 5 had at least 1 episode of acute cellular rejection, 2 sustained a metabolic stroke (with full recovery), and 3 developed mild cardiomyopathy after LT. All have liberalized protein intake, and 9 had no further MDs: median episodes before transplant, 4 (range, 1-30); and median episodes after transplant, 0 (range, 0-5). All survivors made some developmental progress after LT, and none worsened at a median follow-up of 5.8 years (range, 2-23 years). LT in PA significantly reduces the frequency of MDs, can liberalize protein intake and improve quality of life, and should continue to be considered in selected cases.
Copyright © 2019 by the American Association for the Study of Liver Diseases.

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Year:  2020        PMID: 31715057     DOI: 10.1002/lt.25679

Source DB:  PubMed          Journal:  Liver Transpl        ISSN: 1527-6465            Impact factor:   5.799


  2 in total

1.  Therapeutic potential of living donor liver transplantation from heterozygous carrier donors in children with propionic acidemia.

Authors:  Zhi-Gui Zeng; Guang-Peng Zhou; Lin Wei; Wei Qu; Ying Liu; Yu-Le Tan; Jun Wang; Li-Ying Sun; Zhi-Jun Zhu
Journal:  Orphanet J Rare Dis       Date:  2022-02-21       Impact factor: 4.123

2.  Analysis of the relationship between phenotypes and genotypes in 60 Chinese patients with propionic acidemia: a fourteen-year experience at a tertiary hospital.

Authors:  Yi Liu; Zhehui Chen; Hui Dong; Yuan Ding; Ruxuan He; Lulu Kang; Dongxiao Li; Ming Shen; Ying Jin; Yao Zhang; Jinqing Song; Yaping Tian; Yongtong Cao; Desheng Liang; Yanling Yang
Journal:  Orphanet J Rare Dis       Date:  2022-03-24       Impact factor: 4.123

  2 in total

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