Attention-Deficit Hyperactivity Disorder Symptoms in Children and Adolescents with Sex Chromosome Aneuploidy: XXY, XXX, XYY, and XXYY

Attention-Deficit Hyperactivity Disorder Symptoms in Children and Adolescents with Sex Chromosome Aneuploidy: XXY, XXX, XYY, and XXYY
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DOI:
10.1097/dbp.0b013e31824501c8
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发表时间:
2012-05-01
影响因子:
2.4
通讯作者:
Boada, Richard
Boada, Richard
中科院分区:
医学4区
文献类型:
--
作者:
Tartaglia, Nicole R.;Ayari, Natalie;Boada, Richard

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目的:注意力问题、多动和冲动被描述为与性染色体非整倍性(SCA)相关的行为特征。在这项研究中,作者比较了167名年龄在6至20岁之间的参与者的注意力缺陷多动障碍(ADHD)症状,这些参与者患有4种类型的SCA(XXY n = 56,XYY n = 33,XXX n = 25和XXYY n = 53)。他们还评估了与ADHD行为学相关的因素(认知和适应性评分,产前与产后确定),并描述了ADHD治疗患者子集对精神药理学药物的临床反应。研究方法:评估包括病史和发育史,认知和适应功能评估,以及包含DSM-IV标准的家长和教师ADHD问卷。结果如下:在整个研究组中,58%(96/167)在父母报告问卷中符合DSM-IV ADHD标准(XXY为36%,XXX为52%,XYY为76%,XXYY为72%)。注意力不集中亚型在XXY和XXX中最常见,而XYY和XXYY组更可能也有多动/冲动症状。有没有显着差异,在言语,性能,或全量表智商与症状分数在ADHD范围内的儿童相比,低于ADHD范围。然而,与不符合ADHD DSM-IV标准的组相比,在ADHD范围内的组的适应功能评分显著较低。与出生后诊断组相比,产前诊断为XXY的人不太可能符合ADHD的标准。精神药物治疗有效率为78.6%(66/84)。结论:患有SCA的儿童和青少年患ADHD症状的风险增加。建议ADHD的评估和治疗考虑到其他方面的SCA的医疗和行为表型。(J Dev Behav Pediatr 33:309-318,2012)
Objective: Attentional problems, hyperactivity, and impulsivity have been described as behavioral features associated with sex chromosome aneuploidy (SCA). In this study, the authors compare attention-deficit hyperactivity disorder (ADHD) symptoms in 167 participants aged 6 to 20 years with 4 types of SCA (XXY n = 56, XYY n = 33, XXX n = 25, and XXYY n = 53). They also evaluate factors associated with ADHD symptomatology (cognitive and adaptive scores, prenatal vs postnatal ascertainment) and describe the clinical response to psychopharmacologic medications in a subset of patients treated for ADHD. Methods: Evaluation included medical and developmental history, cognitive and adaptive functioning assessment, and parent and teacher ADHD questionnaires containing DSM-IV criteria. Results: In the total study group, 58% (96/167) met DSM-IV criteria for ADHD on parent-report questionnaires (36% in XXY, 52% in XXX, 76% in XYY, and 72% in XXYY). The Inattentive subtype was most common in XXY and XXX, whereas the XYY and XXYY groups were more likely to also have hyperactive/impulsive symptoms. There were no significant differences in Verbal, Performance, or Full Scale IQ between children with symptom scores in the ADHD range compared with those below the ADHD range. However, adaptive functioning scores were significantly lower in the group whose scores in the ADHD range were compared with those of the group who did not meet ADHD DSM-IV criteria. Those with a prenatal diagnosis of XXY were less likely to meet criteria for ADHD compared with the postnatally diagnosed group. Psychopharmacologic treatment with stimulants was effective in 78.6% (66/84). Conclusions: Children and adolescents with SCA are at increased risk for ADHD symptoms. Recommendations for ADHD evaluation and treatment in consideration of other aspects of the SCA medical and behavioral phenotype are provided. (J Dev Behav Pediatr 33:309-318, 2012)