Bonvicini C, Faraone S V, Scassellati C
Genetics Unit, IRCCS Centro S. Giovanni di Dio Fatebenefratelli, Brescia, Italy.
Department of Neuroscience and Physiology, SUNY Upstate Medical University, Syracuse, NY, USA.
Mol Psychiatry. 2016 Jul;21(7):872-84. doi: 10.1038/mp.2016.74. Epub 2016 May 24.
The adult form of attention-deficit/hyperactivity disorder has a prevalence of up to 5% and is the most severe long-term outcome of this common disorder. Family studies in clinical samples as well as twin studies suggest a familial liability and consequently different genes were investigated in association studies. Pharmacotherapy with methylphenidate (MPH) seems to be the first-line treatment of choice in adults with attention-deficit hyperactive disorder (ADHD) and some studies were conducted on the genes influencing the response to this drug. Finally some peripheral biomarkers were identified in ADHD adult patients. We believe this work is the first systematic review and meta-analysis of candidate gene association studies, pharmacogenetic and biochemical (metabolomics) studies performed in adults with ADHD to identify potential genetic, predictive and peripheral markers linked specifically to ADHD in adults. After screening 5129 records, we selected 87 studies of which 61 were available for candidate gene association studies, 5 for pharmacogenetics and 21 for biochemical studies. Of these, 15 genetic, 2 pharmacogenetic and 6 biochemical studies were included in the meta-analyses. We obtained an association between adult ADHD and the gene BAIAP2 (brain-specific angiogenesis inhibitor 1-associated protein 2), even after Bonferroni correction, with any heterogeneity in effect size and no publication bias. If we did not apply the Bonferroni correction, a trend was found for the carriers allele 9R of dopamine transporter SLC6A3 40 bp variable tandem repeat polymorphism (VNTR) and for 6/6 homozygotes of SLC6A3 30 bp VNTR. Negative results were obtained for the 9-6 haplotype, the dopamine receptor DRD4 48 bp VNTR, and the enzyme COMT SNP rs4680. Concerning pharmacogenetic studies, no association was found for the SLC6A3 40 bp and response to MPH with only two studies selected. For the metabolomics studies, no differences between ADHD adults and controls were found for salivary cortisol, whereas lower serum docosahexaenoic acid (DHA) levels were found in ADHD adults. This last association was significant even after Bonferroni correction and in absence of heterogeneity. Other polyunsaturated fatty acids (PUFAs) such as AA (arachidonic acid), EPA (eicosapentaenoic acid) and DyLA (dihomogammalinolenic acid) levels were not different between patients and controls. No publication biases were observed for these markers. Genes linked to dopaminergic, serotoninergic and noradrenergic signaling, metabolism (DBH, TPH1, TPH2, DDC, MAOA, MAOB, BCHE and TH), neurodevelopment (BDNF and others), the SNARE system and other forty genes/proteins related to different pathways were not meta-analyzed due to insufficient data. In conclusion, we found that there were not enough genetic, pharmacogenetic and biochemical studies of ADHD in adults and that more investigations are needed. Moreover we confirmed a significant role of BAIAP2 and DHA in the etiology of ADHD exclusively in adults. Future research should be focused on the replication of these findings and to assess their specificity for ADHD.
成人注意力缺陷多动障碍的患病率高达5%,是这种常见疾病最严重的长期后果。临床样本中的家族研究以及双胞胎研究表明存在家族易感性,因此在关联研究中对不同基因进行了调查。哌甲酯(MPH)药物治疗似乎是成人注意力缺陷多动障碍(ADHD)的一线首选治疗方法,并且针对影响该药物反应的基因开展了一些研究。最后,在ADHD成年患者中确定了一些外周生物标志物。我们认为这项工作是对ADHD成人患者进行的候选基因关联研究、药物遗传学和生化(代谢组学)研究的首次系统评价和荟萃分析,旨在确定与成人ADHD特异性相关的潜在遗传、预测和外周标志物。在筛选了5129条记录后,我们选择了87项研究,其中61项可用于候选基因关联研究,5项用于药物遗传学研究,21项用于生化研究。其中,15项遗传研究、2项药物遗传学研究和6项生化研究纳入了荟萃分析。即使经过Bonferroni校正,我们仍发现成人ADHD与基因BAIAP2(脑特异性血管生成抑制因子1相关蛋白2)之间存在关联,效应大小无任何异质性且无发表偏倚。如果不应用Bonferroni校正,发现多巴胺转运体SLC6A3 40bp可变串联重复多态性(VNTR)的9R等位基因携带者以及SLC6A3 30bp VNTR的6/6纯合子存在一种趋势。9 - 6单倍型、多巴胺受体DRD4 48bp VNTR和酶COMT SNP rs4680的结果为阴性。关于药物遗传学研究,由于仅选择了两项研究,未发现SLC6A3 40bp与对MPH的反应之间存在关联。对于代谢组学研究,ADHD成人与对照组的唾液皮质醇无差异,而ADHD成人的血清二十二碳六烯酸(DHA)水平较低。即使经过Bonferroni校正且无异质性,这一最后的关联仍具有显著性。患者与对照组之间其他多不饱和脂肪酸(PUFA)如花生四烯酸(AA)、二十碳五烯酸(EPA)和二高γ - 亚麻酸(DyLA)水平无差异。这些标志物未观察到发表偏倚。由于数据不足,未对与多巴胺能、血清素能和去甲肾上腺素能信号传导、代谢(DBH、TPH1、TPH2、DDC、MAOA、MAOB、BCHE和TH)、神经发育(BDNF等)、SNARE系统以及与不同途径相关的其他40个基因/蛋白质进行荟萃分析。总之,我们发现针对成人ADHD的遗传、药物遗传学和生化研究不足,需要更多的调查。此外,我们证实了BAIAP2和DHA仅在成人ADHD病因中具有重要作用。未来的研究应侧重于这些发现的重复验证,并评估它们对ADHD的特异性。