Clinically significant psychiatric symptoms among male carriers of the fragile X premutation, with and without FXTAS, and the mediating influence of executive functioning.
Clinically significant psychiatric symptoms among male carriers of the fragile X premutation, with and without FXTAS, and the mediating influence of executive functioning.
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DOI：
10.1080/13854046.2016.1185100
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发表时间：
2016-08
期刊：
影响因子：
--
通讯作者：
Hagerman RJ
中科院分区：
文献类型：
--
作者：
Grigsby J;Brega AG;Bennett RE;Bourgeois JA;Seritan AL;Goodrich GK;Hagerman RJ
阐明脆性X相关震颤/共济失调综合征（FXTAS）的神经精神表型，并评估其由执行障碍综合征介导的程度，执行障碍综合征是该疾病的主要特征。我们研究了脆性X前突变男性携带者与正常等位基因男性相比，有或无FXTAS的临床意义的精神症状的患病率。测量包括神经精神量表（NPI），症状自评量表-90-R（SCL-90-R），和行为控制障碍量表（BDS），一种测量执行功能的方法。组间差异进行了评估，使用逻辑回归，然后通过调解分析与普通最小二乘回归，以评估的贡献，执行障碍综合征观察到的精神领域。患有FXTAS的男性在总体和特定领域表现出更高的临床显著症状率：躯体化、强迫症、抑郁、焦虑、精神病、激动/攻击、冷漠/冷漠、易怒和夜间行为问题。事后分析表明，FXTAS男性患者的精神病可能与参与者对认知和身体功能障碍的准确认识有关，而不是反映精神病。无症状携带者没有表现出临床上显著的精神症状，但当所有携带者与具有正常FMR 1等位基因的男性相比，发现执行功能缺陷介导NPI和SCL-90-R几个领域的得分。基于先前的研究，结果提供了证据表明，男性的精神病表型包括临床上有意义的抑郁症，敌意和易怒，与行为和注意力去抑制有关。这些问题很可能反映了执行功能受损的影响。
To clarify the neuropsychiatric phenotype of fragile X-associated tremor/ataxia syndrome (FXTAS), and assess the extent to which it is mediated by the dysexecutive syndrome that is a major feature of the disorder. We examined the prevalence of clinically meaningful psychiatric symptoms among male carriers of the fragile X premutation, with and without FXTAS, in comparison with men with a normal allele. Measures included the Neuropsychiatric Inventory (NPI), Symptom Checklist-90-R (SCL-90-R), and the Behavioral Dyscontrol Scale (BDS), a measure of executive functioning. Between-group differences were evaluated using logistic regression, followed by a mediation analysis with ordinary least squares regression to assess the contribution of dysexecutive syndrome to the observed psychiatric domains. Men with FXTAS showed higher rates of clinically significant symptoms overall and in specific domains: somatization, obsessive-compulsive, depression, anxiety, psychoticism, agitation/aggression, apathy/indifference, irritability, and nighttime behavior problems. Post hoc analyses suggested that findings of psychoticism among men with FXTAS may be associated with participants’ accurate acknowledgement of cognitive and physical dysfunction, rather than reflecting psychosis. Asymptomatic carriers showed no evidence of clinically significant psychiatric symptoms, but when all carriers were compared with men having a normal FMR1 allele, executive function deficits were found to mediate scores in several domains on both NPI and SCL-90-R. Building on prior research, the results provide evidence that the psychiatric phenotype for men includes clinically meaningful depression, hostility, and irritability, in association with behavioral and attentional disinhibition. It is likely that these problems reflect the effects of impaired executive functioning.
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影响因子：
9.9
作者：
Berger, AK;Fratiglioni, L;Bäckman, L
通讯作者：
Bäckman, L
影响因子：
8.6
作者：
Grigsby, Jim;Brega, Angela G.;Hagerman, Randi J.
通讯作者：
Hagerman, Randi J.
DOI：
10.1002/ajmg.b.30241
发表时间：
2005-11-05
影响因子：
2.8
作者：
Hessl, D;Tassone, F;Hagerman, RJ
通讯作者：
Hagerman, RJ
影响因子：
4.4
作者：
Grigsby, Jim;Brega, Angela G.;Hagerman, Paul J.
通讯作者：
Hagerman, Paul J.
DOI：
10.1097/00004583-199402000-00015
发表时间：
1994-02-01
影响因子：
13.3
作者：
DORN, MB;MAZZOCCO, MMM;HAGERMAN, RJ
通讯作者：
HAGERMAN, RJ