Literature DB >> 29524203

Cognitive and Behavioural Outcomes of Paediatric Liver Transplantation for Ornithine Transcarbamylase Deficiency.

Louise Crowe1,2,3, Vicki Anderson4,5,6,7, Winita Hardikar7,8, Avihu Boneh7,9,10.   

Abstract

Ornithine Trans-Carbamylase (OTC) deficiency is the most common disorder of the urea cycle. Cognitive impairments in skills such as attention and executive function have been reported in individuals with OTC deficiency who are managed with medication. In some cases, children undergo liver transplantation (LTx) to correct the metabolic defect. The metabolic and medical outcomes of LTx are generally good. However, little is known about the impacts on cognition. In this study, four children (three female) completed detailed neuropsychological batteries prior to (n = 6) and following LTx (n = 8 assessments). Children's age at assessment ranged from 3 to 11 years. The battery included standardised, age-referenced measures of intellectual ability (IQ), attention, memory and educational ability. Additionally, parent measures of behaviour and executive function were administered. Generally, there was little change in overall IQ following LTx. Memory and academic skills were at expected levels for the three female patients and gains were made after LTx. Children showed ongoing impairments in attention and parent rated executive function. In conclusion, the immediate effect of LTx on cognition may not appear beneficial in the short-term and impairments in IQ, attention and behaviour persisted after the procedure. However, LTx seems to enable stabilisation to premorbid function in the longer term.

Entities:  

Keywords:  Children; Cognition; Liver transplantation; Ornithine Trans-Carbamylase (OTC) deficiency

Year:  2018        PMID: 29524203      PMCID: PMC6323017          DOI: 10.1007/8904_2018_97

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  4 in total

Review 1.  Executive Functioning in Pediatric Solid Organ Transplant Recipients: A Meta-analytic Review.

Authors:  Grace K Cushman; Mary Gray Stolz; Ronald L Blount; Bonney Reed
Journal:  Transplantation       Date:  2020-02       Impact factor: 5.385

2.  Pre-school neurocognitive and functional outcomes after liver transplant in children with early onset urea cycle disorders, maple syrup urine disease, and propionic acidemia: An inception cohort matched-comparison study.

Authors:  Shailly Jain-Ghai; Ari R Joffe; Gwen Y Bond; Komudi Siriwardena; Alicia Chan; Jason Y K Yap; Morteza Hajihosseini; Irina A Dinu; Bryan V Acton; Charlene M T Robertson
Journal:  JIMD Rep       Date:  2020-01-27

3.  Clinical and cranial MRI features of female patients with ornithine transcarbamylase deficiency: Two case reports.

Authors:  Dan Yu; Guoyan Lu; Rajah Mowshica; Yan Cheng; Fumin Zhao
Journal:  Medicine (Baltimore)       Date:  2019-08       Impact factor: 1.817

4.  Clinical and molecular characteristics of 69 Chinese patients with ornithine transcarbamylase deficiency.

Authors:  Deyun Lu; Feng Han; Wenjuan Qiu; Huiwen Zhang; Jun Ye; Lili Liang; Yu Wang; Wenjun Ji; Xia Zhan; Xuefan Gu; Lianshu Han
Journal:  Orphanet J Rare Dis       Date:  2020-12-03       Impact factor: 4.123

  4 in total

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