Jaquez M, Driscoll D A, Li M, Emanuel B S, Hernandez I, Jaquez F, Lembert N, Ramirez J, Matalon R
Research Institute, Miami Children's Hospital, Florida, USA.
Am J Med Genet. 1997 May 2;70(1):6-10. doi: 10.1002/(sici)1096-8628(19970502)70:1<6::aid-ajmg2>3.0.co;2-z.
We report on an 8-year-old girl with an unbalanced 15;22 translocation and manifestations of DiGeorge syndrome (DGS), velocardiofacial syndrome (VCFS), and other abnormalities. The main manifestations of our patient were feeding difficulties, respiratory infections, short stature, peculiar face with hypertelorism, prominent nose, abnormal ears, microstomia and crowded teeth, short broad neck and shield chest with pectus deformity and widely spaced nipples with abnormal fat distribution, heart defect, scoliosis, asymmetric limb development, abnormal hands and feet, and hyperchromic skin patches. Cytogenetic studies demonstrated a 45,XX,der(15)t(15;22)(p11.2;q11.2), -22 karyotype. Fluorescence in situ hybridization (FISH) studies confirmed loss of the proximal DiGeorge chromosomal region (DGCR). This case adds to the diversity of clinical abnormalities caused by deletions within 22q11.2.
我们报告了一名8岁女孩,她患有不平衡的15;22易位,并伴有迪乔治综合征(DGS)、腭心面综合征(VCFS)及其他异常表现。我们这位患者的主要表现为喂养困难、呼吸道感染、身材矮小、面容奇特,表现为眼距增宽、鼻子突出、耳朵异常、小口畸形和牙齿拥挤、短而宽的颈部、盾状胸伴胸壁畸形以及乳头间距增宽且脂肪分布异常、心脏缺陷、脊柱侧弯、肢体发育不对称、手足异常以及皮肤色素沉着斑。细胞遗传学研究显示核型为45,XX,der(15)t(15;22)(p11.2;q11.2), -22。荧光原位杂交(FISH)研究证实了近端迪乔治染色体区域(DGCR)的缺失。该病例增加了由22q11.2内缺失导致的临床异常的多样性。