Poirsier Celine, Landais Emilie, Bednarek Nathalie, Nobecourt Jean-Marie, Khoury Maroun, Schmidt Pascal, Morville Patrice, Gruson Nadine, Clomes Sandrine, Michel Nicole, Riot Anita, Manjeongean Christelle, Gaillard Dominique, Doco-Fenzy Martine
Department of Genetics, Robert Debre University Hospital, Reims, France.
Department of Genetics, Robert Debre University Hospital, Reims, France.
Eur J Med Genet. 2014 Apr;57(5):185-94. doi: 10.1016/j.ejmg.2013.12.009. Epub 2014 Feb 3.
The duplication of the short arm (p) of chromosome 12 is a rare chromosomal abnormality, and most reported cases result from malsegregation of a balanced parental translocation associated with other chromosomal imbalances. Of the reported cases, only 15 involve a pure and complete 12p duplication and only 10 involve a pure and partial duplication overlapping the 12p12.3p13.1 region, including a single instance of an inherited duplication in two related individuals. Here, we report three new patients with a pure 12p duplication, detected by conventional cytogenetic studies and characterized by array-comparative genomic hybridization (array-CGH) and fluorescence in situ hybridization (FISH). The first patient was a child carrying a de novo inverted duplication of the short arm of chromosome 12. His phenotype was similar to that of the "trisomy 12p syndrome", characterized by developmental delays and craniofacial abnormalities including a high forehead, a short nose with anteverted nostrils and an everted lower lip. The second and third patients were a mother and son with a direct 12p12.3p13.1 duplication, exhibiting a milder phenotype characterized by moderate developmental delays, dysmorphic facial features, behavioral problems and obesity. The present data, including the rarity of the familial cases, should contribute to our knowledge of the genotype/phenotype correlation in trisomy 12p patients.
12号染色体短臂(p)重复是一种罕见的染色体异常,大多数报道的病例是由与其他染色体失衡相关的平衡亲本易位错误分离所致。在已报道的病例中,仅有15例涉及纯合且完整的12p重复,仅有10例涉及与12p12.3 - p13.1区域重叠的纯合且部分重复,其中包括两个相关个体中单一的遗传性重复实例。在此,我们报告3例新的纯合12p重复患者,通过传统细胞遗传学研究检测到,并通过阵列比较基因组杂交(array - CGH)和荧光原位杂交(FISH)进行特征分析。首例患者是一名儿童,携带12号染色体短臂的新发倒位重复。他的表型与“12p三体综合征”相似,特征为发育迟缓及颅面异常,包括额头高、鼻孔前倾的短鼻和下唇外翻。第二例和第三例患者是一对母子,携带直接的12p12.3 - p13.1重复,表现出较轻的表型,特征为中度发育迟缓、面部畸形特征、行为问题和肥胖。包括家族性病例罕见性在内的现有数据,应有助于我们了解12p三体患者的基因型/表型相关性。