Department of Psychiatry, Rudolf Magnus Institute of Neuroscience, University Medical Center Utrecht, Utrecht, The Netherlands; Dalglish Family Hearts and Minds Clinic for 22q11.2 Deletion Syndrome, Toronto General Hospital, University Health Network, Toronto, Ontario, Canada; Clinical Genetics Research Program, Centre for Addiction and Mental Health, Toronto, Ontario, Canada.
Department of Psychiatry, Rudolf Magnus Institute of Neuroscience, University Medical Center Utrecht, Utrecht, The Netherlands; Dalglish Family Hearts and Minds Clinic for 22q11.2 Deletion Syndrome, Toronto General Hospital, University Health Network, Toronto, Ontario, Canada; Clinical Genetics Research Program, Centre for Addiction and Mental Health, Toronto, Ontario, Canada.
Schizophr Res. 2017 Oct;188:59-62. doi: 10.1016/j.schres.2017.01.032. Epub 2017 Jan 21.
Individuals with 22q11.2 deletion syndrome (22q11DS) have a 25% risk for schizophrenia and related psychotic disorders. Some have hypothesized that Autism Spectrum Disorders (ASDs) diagnosed in children with 22q11DS may actually represent the social-communicative defects often observed during the early developmental stages of schizophrenia.
We prospectively studied 89 children with 22q11DS to test this hypothesis. At baseline, the Autism Diagnostic Interview was used to assess ASD, evaluating both current and early childhood behaviors. At follow-up, the Schedule for Affective Disorders and Schizophrenia for School-Age Children (K-SADS) was used to determine development of a psychotic disorder or psychotic symptoms.
The average age (±SD) at first and last assessments was 14.3±1.9 and 19.0±3.0years, respectively. Nineteen (21.3%) children developed a psychotic disorder. Contrary to our hypothesis, there was no significant difference in the proportion that developed a psychotic disorder, comparing those with (n=9, 17.3%) and those without ASD at baseline (n=10, 27%; OR=0.500, 95% CI=0.160-1.569, p=0.235). Similar results were obtained using autistic symptom severity as quantitative predicting variable, psychotic symptoms as the outcome, and when correcting for age, gender and full scale IQ.
Results indicate that in children with 22q11DS, early childhood autistic features are not associated with an increased risk for subsequent development of psychotic disorders or symptoms, replicating previous retrospective findings in adults with 22q11DS. These results indicate that ASD and psychotic disorders can emerge independently, as pleiotropic phenotypes in the context of 22q11DS.
患有 22q11.2 缺失综合征(22q11DS)的个体患精神分裂症和相关精神病性障碍的风险为 25%。一些人假设,在患有 22q11DS 的儿童中诊断出的自闭症谱系障碍(ASD)实际上可能代表精神分裂症早期发育阶段经常观察到的社会交流缺陷。
我们前瞻性研究了 89 名患有 22q11DS 的儿童,以检验这一假设。在基线时,使用自闭症诊断访谈来评估 ASD,评估当前和儿童早期的行为。在随访时,使用儿童青少年情感障碍和精神分裂症的时间表(K-SADS)来确定精神病性障碍或精神病性症状的发展。
首次和最后一次评估的平均年龄(±SD)分别为 14.3±1.9 和 19.0±3.0 岁。19 名(21.3%)儿童患上了精神病性障碍。与我们的假设相反,在基线时患有 ASD 的儿童(n=9,17.3%)和没有 ASD 的儿童(n=10,27%)中,发展为精神病性障碍的比例没有显著差异(OR=0.500,95%CI=0.160-1.569,p=0.235)。使用作为定量预测变量的自闭症症状严重程度、作为结果的精神病症状,以及在校正年龄、性别和全量表智商后,也得到了相似的结果。
结果表明,在患有 22q11DS 的儿童中,儿童早期的自闭症特征与随后发展为精神病性障碍或症状的风险增加无关,这与之前在患有 22q11DS 的成年人中进行的回顾性研究结果相吻合。这些结果表明,在 22q11DS 背景下,ASD 和精神病性障碍可以作为多效表型独立出现。