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鸟氨酸转氨甲酰酶缺乏症小儿肝移植的认知和行为结果

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

作者信息

Crowe Louise, Anderson Vicki, Hardikar Winita, Boneh Avihu

机构信息

School of Psychological Sciences, University of Melbourne, Parkville, VIC, Australia.

Child Neuropsychology, Murdoch Children's Research Institute, Melbourne, VIC, Australia.

出版信息

JIMD Rep. 2019;43:19-25. doi: 10.1007/8904_2018_97. Epub 2018 Mar 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.

摘要

鸟氨酸转氨甲酰酶(OTC)缺乏症是尿素循环中最常见的疾病。据报道,接受药物治疗的OTC缺乏症患者存在注意力和执行功能等技能方面的认知障碍。在某些情况下,儿童会接受肝移植(LTx)来纠正代谢缺陷。肝移植的代谢和医疗结果总体良好。然而,对于其对认知的影响却知之甚少。在本研究中,四名儿童(三名女性)在肝移植前(n = 6次评估)和肝移植后(n = 8次评估)完成了详细的神经心理测试。评估时儿童的年龄在3至11岁之间。测试包括标准化的、与年龄相关的智力能力(IQ)、注意力、记忆力和教育能力测量。此外,还进行了家长对行为和执行功能的测量。总体而言,肝移植后总体智商变化不大。三名女性患者的记忆力和学业技能处于预期水平,且肝移植后有所提高。儿童在注意力方面持续存在障碍,家长评定的执行功能也存在问题。总之,肝移植对认知的直接影响在短期内可能并不明显有益,术后智商、注意力和行为方面的障碍仍然存在。然而,从长期来看,肝移植似乎能够使功能稳定到病前水平。

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