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大龄儿童及青少年的帕利斯特-基利安综合征

Pallister-Killian syndrome in older children and adolescents.

作者信息

Horneff G, Majewski F, Hildebrand B, Voit T, Lenard H G

机构信息

Department of Pediatrics, University of Düsseldorf, Germany.

出版信息

Pediatr Neurol. 1993 Jul-Aug;9(4):312-5. doi: 10.1016/0887-8994(93)90071-j.

Abstract

The Pallister-Killian syndrome is caused by a mosaic tetrasomy of the short arm of chromosome 12. Although analysis of peripheral blood lymphocytes usually reveals a normal karyotype, an isochromosome 12p mosaicism is detectable in fibroblast cultures; therefore, in this rare chromosomal aberration, clinical recognition is crucial for appropriate cytogenetic investigations. The phenotype of younger children has already been well documented. During childhood and adolescence, however, the phenotype changes markedly. The disorder in older children and young adults is characterized by a coarse and flat facies, macroglossia prognathia, everted lower lip, and severe psychomotor retardation with muscular hypertonia and contractures. Two severely mentally retarded patients are reported whose diagnoses were confirmed by fibroblast cultures at ages 16 and 21 years.

摘要

帕利斯特-基利安综合征由12号染色体短臂的嵌合四体性引起。虽然外周血淋巴细胞分析通常显示核型正常,但在成纤维细胞培养物中可检测到12p等臂染色体嵌合体;因此,在这种罕见的染色体畸变中,临床识别对于适当的细胞遗传学检查至关重要。年幼儿童的表型已有充分记录。然而,在儿童期和青春期,表型会发生明显变化。大龄儿童和青年成人的疾病特征为面容粗糙扁平、巨舌、突颌、下唇外翻,以及伴有肌肉张力亢进和挛缩的严重精神运动发育迟缓。报告了两名严重智力发育迟缓的患者,其诊断在16岁和21岁时通过成纤维细胞培养得到证实。

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