Department of Neurology, Wake Forest School of Medicine, Wake Forest Baptist Health, Winston-Salem, NC, USA.
Neurology. 2012 Sep 11;79(11):1168-73. doi: 10.1212/WNL.0b013e3182698d6c. Epub 2012 Aug 29.
Rapid-onset dystonia-parkinsonism (RDP) is caused by a variety of missense mutations in the ATP1A3 gene. Psychiatric comorbidity has been reported, although systematic examination of psychiatric disease in individuals with RDP is lacking. This study examines psychiatric morbidity for 23 patients with RDP in 10 families with family member control subjects and in 3 unrelated patients with RDP, totaling 56 individuals.
Twenty-nine ATP1A3 mutation-positive individuals were examined; 26 exhibited motor symptoms (motor manifesting carrier [MMC]) and 3 did not (nonmotor manifesting carriers [NMC]). Twenty-seven ATP1A3 mutation-negative participants (noncarriers [NC]) were included. Rates of psychiatric illness for patients with RDP and related asymptomatic gene mutation carriers were compared with those for related nonmutation carriers. Outcome measures included the Unified Parkinson's Disease Rating Scale, Burke-Fahn-Marsden Dystonia Rating Scale, Instrumental Activities of Daily Living, Composite International Diagnostic Interview, Structured Clinical Interview for DSM-IV, Hamilton Depression Scale, Hamilton Anxiety Scale, and Yale-Brown Obsessive-Compulsive Scale.
NMC participants did not report any history of psychiatric disorder. Findings in MMC and NC groups included anxiety (MMC 48, NC 41%), mood (MMC 50%, NC 22%), psychotic (MMC 19%, NC 0%), and substance abuse/dependence (MMC 38%, NC 27%).
ATP1A3 mutations cause a wide spectrum of motor and nonmotor features. Psychotic symptoms tended to emerge before or concurrent with motor symptom onset, suggesting that this could be another expression of the ATP1A3 gene mutation.
快速进展性肌张力障碍-帕金森病(RDP)是由 ATP1A3 基因的多种错义突变引起的。虽然已经报道了精神共病,但缺乏对 RDP 个体精神疾病的系统检查。本研究检查了 10 个家族的 23 名 RDP 患者、10 个家族的家庭成员对照和 3 名无相关 RDP 的患者(共 56 人)的精神发病率。
共检查了 29 名 ATP1A3 突变阳性个体,其中 26 名表现为运动症状(运动显性携带者[MMC]),3 名不表现(非运动显性携带者[NMC])。共纳入 27 名 ATP1A3 突变阴性参与者(非携带者[NC])。比较 RDP 患者和相关无症状基因突变携带者与相关非突变携带者的精神疾病发病率。评估指标包括统一帕金森病评定量表、Burke-Fahn-Marsden 肌张力障碍评定量表、日常生活活动能力量表、复合国际诊断访谈、DSM-IV 结构化临床访谈、汉密尔顿抑郁量表、汉密尔顿焦虑量表和耶鲁-布朗强迫量表。
NMC 参与者未报告任何精神疾病史。MMC 和 NC 组的发现包括焦虑(MMC 48%,NC 41%)、情绪(MMC 50%,NC 22%)、精神病(MMC 19%,NC 0%)和物质滥用/依赖(MMC 38%,NC 27%)。
ATP1A3 突变引起广泛的运动和非运动特征。精神病症状往往在运动症状出现之前或同时出现,表明这可能是 ATP1A3 基因突变的另一种表现。