Horn D, Majewski F, Hildebrandt B, Körner H
Institute of Medical Genetics, School of Medicine (Charité), Humboldt University, Berlin, Germany.
J Med Genet. 1995 Jan;32(1):68-71. doi: 10.1136/jmg.32.1.68.
We report on two patients with Pallister-Killian syndrome: an 18 month old male infant followed since the neonatal period and a 4 year old boy. Prenatal diagnosis by chorionic villi sampling (CVS) in the first case showed a normal karyotype without mosaicism. Chromosome analysis on peripheral lymphocytes of the newborn also showed a normal karyotype. The clinical diagnosis of Pallister-Killian syndrome was made after the first year of life because of the typical facial dysmorphism and other characteristic clinical features, such as frontotemporal alopecia, depigmented area of the skin, sensorineural hearing loss, and severe psychomotor retardation. Chromosome analysis from skin fibroblasts now showed an isochromosome 12p mosaicism. The origin of the extra chromosome was confirmed by in situ hybridisation using a chromosome 12 specific library. In the second case chromosomal analysis from peripheral lymphocytes at the age of 19 months showed a normal karyotype 46,XY. Following the clinical diagnosis of Pallister-Killian syndrome a superficial skin biopsy was performed which showed very poor and slow growth of cells and a normal karyotype. Because of the typical symptoms a larger and deeper skin biopsy was performed from which there was rapid growth of fibroblasts. Now the diagnosis was established on the basis of the presence of an i(12p) extra chromosome in 69% of the metaphases.
我们报告了两名患有帕利斯特-基利安综合征的患者:一名自新生儿期起就接受随访的18个月大男婴和一名4岁男孩。第一例通过绒毛取样(CVS)进行的产前诊断显示核型正常,无嵌合体现象。新生儿外周淋巴细胞的染色体分析也显示核型正常。由于典型的面部畸形及其他特征性临床症状,如额颞部脱发、皮肤色素脱失区、感音神经性听力损失和严重精神运动发育迟缓,在患儿1岁后做出了帕利斯特-基利安综合征的临床诊断。此时皮肤成纤维细胞的染色体分析显示存在12号染色体短臂等臂染色体嵌合体。使用12号染色体特异性文库进行原位杂交证实了额外染色体的来源。在第二例中,19个月大时外周淋巴细胞的染色体分析显示核型正常,为46,XY。在帕利斯特-基利安综合征临床诊断后,进行了一次浅表皮肤活检,结果显示细胞生长非常缓慢且不良,核型正常。由于出现了典型症状,于是进行了一次更大且更深部的皮肤活检,从中发现成纤维细胞生长迅速。现在根据69%的中期相中存在一条额外的12号染色体短臂等臂染色体确诊。