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

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

Journal

JIMD reports
ISSN: 2192-8304
Titre abrégé: JIMD Rep
Pays: United States
ID NLM: 101568557

Informations de publication

Date de publication:
2019
Historique:
received: 01 10 2017
accepted: 12 02 2018
revised: 09 02 2018
pubmed: 11 3 2018
medline: 11 3 2018
entrez: 11 3 2018
Statut: ppublish

Résumé

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.

Identifiants

pubmed: 29524203
doi: 10.1007/8904_2018_97
pmc: PMC6323017
doi:

Types de publication

Journal Article

Langues

eng

Pagination

19-25

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Auteurs

Louise Crowe (L)

School of Psychological Sciences, University of Melbourne, Parkville, VIC, Australia. louise.crowe@mcri.edu.au.
Child Neuropsychology, Murdoch Children's Research Institute, Melbourne, VIC, Australia. louise.crowe@mcri.edu.au.
Department of Psychology, Royal Children's Hospital, Melbourne, VIC, Australia. louise.crowe@mcri.edu.au.

Vicki Anderson (V)

School of Psychological Sciences, University of Melbourne, Parkville, VIC, Australia.
Child Neuropsychology, Murdoch Children's Research Institute, Melbourne, VIC, Australia.
Department of Psychology, Royal Children's Hospital, Melbourne, VIC, Australia.
Department of Paediatrics, University of Melbourne, Parkville, VIC, Australia.

Winita Hardikar (W)

Department of Paediatrics, University of Melbourne, Parkville, VIC, Australia.
Department of Gastroenterology and Nutrition, Royal Children's Hospital, Melbourne, VIC, Australia.

Avihu Boneh (A)

Department of Paediatrics, University of Melbourne, Parkville, VIC, Australia.
Metabolic Research, Murdoch Children's Research Institute, Royal Children's Hospital, Melbourne, VIC, Australia.
Department of Metabolic Medicine, Royal Children's Hospital, Melbourne, VIC, Australia.

Classifications MeSH