Ben-Abdallah-Bouhjar Inesse, Hannachi Hanene, Labalme Audrey, Gmidène Abir, Mougou Soumaya, Soyah Najla, Gribaa Moez, Sanlaville Damien, Elghezal Hatem, Saad Ali
Cytogenetics and Reproductive Biology Department, Farhat Hached University Teaching Hospital, Avenue Ibn Eljazzar, 4000 Sousse, Tunisia.
Eur J Med Genet. 2012 Aug-Sep;55(8-9):461-5. doi: 10.1016/j.ejmg.2012.04.011. Epub 2012 Jun 7.
Duplications of the long arm of the X chromosome are rare. The infantile phenotype shares some resemblance with the Prader-Willi syndrome, presenting severe psychomotor retardation, facial dysmorphic features with a broad face, a small mouth and a thin pointed nose, hypotonia, urogenital malformation and proneness to infections. We report a boy with an additional Xq27-qter chromosome segment translocated onto the short arm of chromosome 3. The karyotype was 46,XY,der(3)t(X;3)(q27.3; p26.3)mat. This cryptic unbalanced X-autosome translocation resulted in Xq27-qter functional disomy and a deletion 3p26.3. A detailed analysis of the constitutional chromosomal changes in the patient was performed using array-CGH, FISH and PCR. The aim was to characterize the size and the location of the duplication Xq27-qter (8.18 Mb) and of the deletion 3p26.3 (1.05 Mb), to establish phenotype-genotype correlations and to offer genetic counselling.
X染色体长臂重复较为罕见。婴儿期表型与普拉德-威利综合征有一些相似之处,表现为严重的精神运动发育迟缓、面部畸形特征,如脸宽、小嘴和尖细鼻子、肌张力减退、泌尿生殖系统畸形以及易感染。我们报告了一名男孩,其额外的Xq27-qter染色体片段易位到3号染色体短臂上。核型为46,XY,der(3)t(X;3)(q27.3; p26.3)mat。这种隐匿的不平衡X-常染色体易位导致Xq27-qter功能二体性和3p26.3缺失。使用阵列比较基因组杂交(array-CGH)、荧光原位杂交(FISH)和聚合酶链反应(PCR)对患者的染色体结构变化进行了详细分析。目的是确定重复的Xq27-qter(8.18 Mb)和缺失的3p26.3(1.05 Mb)的大小和位置,建立表型-基因型相关性并提供遗传咨询。