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早期治疗苯丙酮尿症患者的社会认知功能和社交技能:一项苯丙酮尿症-COBESO研究

Social-cognitive functioning and social skills in patients with early treated phenylketonuria: a PKU-COBESO study.

作者信息

Jahja Rianne, van Spronsen Francjan J, de Sonneville Leo M J, van der Meere Jaap J, Bosch Annet M, Hollak Carla E M, Rubio-Gozalbo M Estela, Brouwers Martijn C G J, Hofstede Floris C, de Vries Maaike C, Janssen Mirian C H, van der Ploeg Ans T, Langendonk Janneke G, Huijbregts Stephan C J

机构信息

University of Groningen, University Medical Center Groningen, Beatrix Children's Hospital, Division of Metabolic Diseases CA33, PO Box 30.001, 9700 RB, Groningen, The Netherlands.

Department of Clinical Child and Adolescent Studies & Leiden Institute for Brain and Cognition, Leiden University, Leiden, The Netherlands.

出版信息

J Inherit Metab Dis. 2016 May;39(3):355-362. doi: 10.1007/s10545-016-9918-0. Epub 2016 Feb 25.

Abstract

OBJECTIVE

Early treatment of phenylketonuria (ET-PKU) prevents mental retardation, but many patients still show cognitive and mood problems. In this study, it was investigated whether ET-PKU-patients have specific phenylalanine (Phe-)related problems with respect to social-cognitive functioning and social skills.

METHODS

Ninety five PKU-patients (mean age 21.6 ± 10.2 years) and 95 healthy controls (mean age 19.6 ± 8.7 years) were compared on performance of computerized and paper-and-pencil tasks measuring social-cognitive abilities and on parent- and self-reported social skills, using multivariate analyses of variance, and controlling for general cognitive ability (IQ-estimate). Further comparisons were made between patients using tetrahydrobiopterin (BH4, N = 30) and patients not using BH4. Associations with Phe-levels on the day of testing, during childhood, during adolescence and throughout life were examined.

RESULTS

PKU-patients showed poorer social-cognitive functioning and reportedly had poorer social skills than controls (regardless of general cognitive abilities). Quality of social-cognitive functioning was negatively related to recent Phe-levels and Phe-levels between 8 and 12 years for adolescents with PKU. Quality of social skills was negatively related to lifetime phenylalanine levels in adult patients, and specifically to Phe-levels between 0 and 7, and between 8 and 12 years. There were no differences with respect to social outcome measures between the BH4 and non-BH4 groups.

CONCLUSION

PKU-patients have Phe-related difficulties with social-cognitive functioning and social skills. Problems seem to be more evident among adolescents and adults with PKU. High Phe-levels during childhood and early adolescence seem to be of greater influence than current and recent Phe-levels for these patients.

摘要

目的

苯丙酮尿症的早期治疗(ET-PKU)可预防智力发育迟缓,但许多患者仍存在认知和情绪问题。本研究调查了ET-PKU患者在社会认知功能和社交技能方面是否存在特定的与苯丙氨酸(Phe-)相关的问题。

方法

采用多变量方差分析,并控制一般认知能力(智商估计值),比较了95名PKU患者(平均年龄21.6±10.2岁)和95名健康对照者(平均年龄19.6±8.7岁)在测量社会认知能力的计算机化和纸笔任务中的表现,以及父母报告和自我报告的社交技能。对使用四氢生物蝶呤(BH4,N = 30)的患者和未使用BH4的患者进行了进一步比较。研究了测试当天、儿童期、青春期和一生中Phe水平的相关性。

结果

PKU患者的社会认知功能较差,据报道其社交技能也比对照组差(无论一般认知能力如何)。PKU青少年的社会认知功能质量与近期Phe水平以及8至12岁之间的Phe水平呈负相关。成年患者的社交技能质量与终生苯丙氨酸水平呈负相关,特别是与0至7岁以及8至12岁之间的Phe水平呈负相关。BH4组和非BH4组在社会结局指标方面没有差异。

结论

PKU患者在社会认知功能和社交技能方面存在与Phe相关的困难。这些问题在PKU青少年和成年人中似乎更为明显。对于这些患者来说,儿童期和青春期早期的高Phe水平似乎比当前和近期的Phe水平影响更大。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/ed66/4851698/24c56dcdf88b/10545_2016_9918_Fig1_HTML.jpg

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