Department of Psychiatry, Center for Genetic Disorders of Cognition and Behavior, Kennedy Krieger Institute, Baltimore, Maryland 21205, USA.
Am J Med Genet B Neuropsychiatr Genet. 2011 Jan;156B(1):59-66. doi: 10.1002/ajmg.b.31138. Epub 2010 Nov 2.
Nail-Patella syndrome (NPS) is an autosomal dominant disorder that is the result of heterozygous loss-of-function mutations in LMX1B, coding for a LIM homeobox (LIM-HD) transcription factor. Analyses of lmx1b mutant mice have revealed the role of Lmx1b in the development of mesencephalic dopaminergic neurons and the serotonergic system; these areas have been linked with symptoms of attention deficit hyperactivity disorder (ADHD) and major depressive disorder (MDD). Fifty adults (38 females, 12 males) with NPS completed the Conners' Adult ADHD Rating Scales-Self-report: Long Version (CAARS) and Beck Depression Inventory-II (BDI-II). The objective was to describe the neurobehavioral phenotype of these subjects and examine possible relationships between neurobehavioral symptoms and NPS. Elevated levels of DSM-IV-TR ADHD Inattentive symptoms were reported on the CAARS by 22% of the NPS sample. The BDI-II Total score was elevated for 40% of the NPS sample. There was a significant increase in the odds of an elevated BDI-II Total score when any of the three CAARS scales were elevated (odds ratios ranging from 11.455 to 15.615). The CAARS and BDI-II did not significantly differ with gender, age, or education level. There was no significant association between genetic mutation-predicted protein status and elevations on CAARS or BDI-II. Individuals with NPS reported co-occurring symptoms of ADHD and MDD, with higher levels of co-occurrence than reported in the literature for the general population. The co-occurrence of these symptoms may be related to mesencephalic dopaminergic neurologic pathway abnormalities that are a consequence of LMX1B loss of function.
指甲髌骨综合征(NPS)是一种常染色体显性遗传疾病,其病因是 LMX1B 基因杂合性功能丧失突变,该基因编码 LIM 同源盒(LIM-HD)转录因子。对 lmx1b 突变小鼠的分析揭示了 Lmx1b 在中脑多巴胺能神经元和 5-羟色胺能系统发育中的作用;这些区域与注意缺陷多动障碍(ADHD)和重度抑郁症(MDD)的症状有关。50 名 NPS 成年患者(38 名女性,12 名男性)完成了 Conners 成人 ADHD 评定量表-自陈式:长版(CAARS)和贝克抑郁量表-II(BDI-II)。目的是描述这些患者的神经行为表型,并探讨神经行为症状与 NPS 之间的可能关系。22%的 NPS 样本在 CAARS 上报告存在 DSM-IV-TR ADHD 注意力不集中症状。40%的 NPS 样本 BDI-II 总分升高。当 CAARS 中的任何三个量表升高时,BDI-II 总分升高的可能性显著增加(比值比范围为 11.455 至 15.615)。CAARS 和 BDI-II 与性别、年龄或教育水平无关。CAARS 或 BDI-II 升高与遗传突变预测的蛋白状态之间没有显著关联。NPS 患者报告存在 ADHD 和 MDD 的共病症状,其共病发生率高于文献中一般人群的报道。这些症状的共病可能与 LMX1B 功能丧失导致的中脑多巴胺能神经通路异常有关。