Yan W, Jacobsen L K, Krasnewich D M, Guan X Y, Lenane M C, Paul S P, Dalwadi H N, Zhang H, Long R T, Kumra S, Martin B M, Scambler P J, Trent J M, Sidransky E, Ginns E I, Rapoport J L
Child Psychiatry Branch, National Institute of Mental Health, Bethesda, Maryland 20892-4405, USA.
Am J Med Genet. 1998 Feb 7;81(1):41-3.
Since its first description almost a century ago schizophrenia with childhood onset, a rare yet devastating disorder, has been diagnosed in children as young as age 5. Recently, the velocardiofacial syndrome, whose underlying cause is interstitial deletions of 22q11.2, was found in 2 of 100 cases of schizophrenics with adult onset [Karayiorgou et al., Proc Natl Acad Sci USA 92: 7612-7616, 1995]. No study has documented the prevalence of velocardiofacial syndrome and the 22q11.2 deletion in a population of schizophrenics with childhood onset. Here we describe the result of such a study in a sample originally selected for a trial of atypical antipsychotic drugs. A separate group of patients was also included in the study; they can best be accounted for as a variant of childhood-onset schizophrenia (COS) and had been provisionally termed "multidimensionally impaired." Fluorescent in situ hybridization screening of 32 COS and 21 multidimensionally impaired patients revealed 1 COS patient with an interstitial deletion spanning at least 2.5 megabases.
自近一个世纪前首次被描述以来,儿童期起病的精神分裂症是一种罕见但极具破坏性的疾病,在年仅5岁的儿童中就已被诊断出来。最近,在100例成人起病的精神分裂症患者中,有2例被发现患有心脏颜面综合征,其潜在病因是22q11.2间质性缺失[卡拉约尔古等人,《美国国家科学院院刊》92: 7612 - 7616,1995]。尚无研究记录心脏颜面综合征和22q11.2缺失在儿童期起病的精神分裂症患者群体中的患病率。在此,我们描述了在最初为非典型抗精神病药物试验而选取的样本中进行此类研究的结果。另一组患者也被纳入该研究;他们最适合被视为儿童期起病的精神分裂症(COS)的一种变体,并且曾被临时称为“多维度受损”。对32例COS患者和21例多维度受损患者进行荧光原位杂交筛查发现,有1例COS患者存在跨度至少为2.5兆碱基的间质性缺失。