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一名具有畸形特征、智力残疾和严重全身性癫痫患者21号染色体间质缺失(21q22.13q22.3)的分子细胞遗传学特征

Molecular cytogenetic characterization of an interstitial deletion of chromosome 21 (21q22.13q22.3) in a patient with dysmorphic features, intellectual disability and severe generalized epilepsy.

作者信息

Valetto Angelo, Orsini Alessandro, Bertini Veronica, Toschi Benedetta, Bonuccelli Alice, Simi Francesca, Sammartino Irene, Taddeucci Grazia, Simi Paolo, Saggese Giuseppe

机构信息

Cytogenetics and Molecular Genetic Unit, A.O.U. Pisana, Ospedale S. Chiara, via Roma 57, Pisa, Italy.

出版信息

Eur J Med Genet. 2012 May;55(5):362-6. doi: 10.1016/j.ejmg.2012.03.011. Epub 2012 Apr 24.

DOI:10.1016/j.ejmg.2012.03.011
PMID:22548977
Abstract

We report on a de novo interstitial deletion of chromosome 21q in a patient presenting with characteristic facial features, intellectual disability, and epilepsy. The deletion extent was about 4.9 Mb from position 37713441 bp (21q22.13) to position 42665162 bp (21q22.3) (NCBI36/hg18 map). Patients with partial monosomy 21 are quite rare; this anomaly has been associated with a wide spectrum of clinical signs, ranging from very mild to quite severe phenotypes. This variability results from variability in the deleted regions, thus accurate molecular definition of the chromosomal breakpoints is necessary to make better genotype-phenotype correlations. We compared our patient's phenotype with the few other patients reported in the literature and found to have similar deletion when analyzed by array CGH. The minimal overlapping region contains only two genes, DYRK1A and KCNJ6, which may play a major role in these patients' phenotype.

摘要

我们报告了一例21号染色体q臂间质性从头缺失的患者,该患者具有典型的面部特征、智力残疾和癫痫症状。缺失范围约为4.9兆碱基对,从37713441碱基对位置(21q22.13)到42665162碱基对位置(21q22.3)(NCBI36/hg18图谱)。21号染色体部分单体患者非常罕见;这种异常与广泛的临床体征相关,从非常轻微到相当严重的表型不等。这种变异性源于缺失区域的变异性,因此准确分子定义染色体断点对于更好地建立基因型-表型相关性是必要的。我们将我们患者的表型与文献中报道的其他少数患者进行了比较,通过阵列比较基因组杂交分析发现具有相似的缺失。最小重叠区域仅包含两个基因,DYRK1A和KCNJ6,它们可能在这些患者的表型中起主要作用。

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