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22q11.2 缺失综合征患者挪威全国调查中年龄相关的临床问题。

Age-dependent clinical problems in a Norwegian national survey of patients with the 22q11.2 deletion syndrome.

机构信息

Department of Endocrinology, Division of Medicine, Akershus University Hospital, Sykehusveien 27, 1478 Lørenskog, Norway.

出版信息

Eur J Pediatr. 2010 Aug;169(8):983-9. doi: 10.1007/s00431-010-1161-3. Epub 2010 Feb 26.

Abstract

Patients with the 22q11.2 deletion syndrome display a wide phenotypic variation that is important for clinical follow-up. In this national survey of 60 patients (ages 1 to 54 years) diagnosed by Fluorescence in situ hybridization test, data were collected from medical records, a physical examination, and a semistructured interview. Ultrasound investigation of the kidneys was also performed. In addition, multiplex ligation probe amplification assay was performed to detect deletion size. Phenotypic features leading to the genetic diagnosis were noted. The patients showed a variety of organ malformations including 39 with heart anomalies. Only 20 individuals had been diagnosed with 22q11.2 DS in the first year of life. Four patients had renal and five males had genital malformations. The increased infection susceptibility (excluding otitis media) and most feeding difficulties subsided during early childhood. Speech difficulties started early and were a major problem for many patients at least until 10 years of age. Ten patients developed kyphoscoliosis in late childhood. In teenagers and adults, abnormal social behavior, learning disabilities, and psychiatric symptoms dominated. Our study which also includes adult patients emphasizes a marked change in challenges in individuals with the 22q11.2 deletion syndrome with increasing age.

摘要

患有 22q11.2 缺失综合征的患者表现出广泛的表型变异,这对于临床随访很重要。在这项针对 60 名经荧光原位杂交试验诊断的患者(年龄 1 至 54 岁)的全国性调查中,从病历、体检和半结构化访谈中收集数据。还对肾脏进行了超声检查。此外,还进行了多重连接探针扩增检测,以检测缺失大小。记录导致遗传诊断的表型特征。患者表现出多种器官畸形,包括 39 例心脏异常。只有 20 人在生命的第一年被诊断出患有 22q11.2 DS。4 名患者有肾脏畸形,5 名男性有生殖器畸形。感染易感性(不包括中耳炎)增加和大多数喂养困难在幼儿期得到缓解。言语困难很早就开始了,至少在 10 岁之前,许多患者都存在这个问题。10 名患者在儿童后期发展为脊柱侧凸。在青少年和成年人中,异常的社会行为、学习障碍和精神症状占主导地位。我们的研究还包括成年患者,强调了随着年龄的增长,22q11.2 缺失综合征患者的挑战发生了明显变化。

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