Alao M J, Lalèyè A, Lalya F, Hans Ch, Abramovicz M, Morice-Picard F, Arveiler B, Lacombe D, Rooryck C
Service de Pédiatrie, Hôpital de la Mère et de l'Enfant Lagune, 01 BP 107, Cotonou, Benin.
Eur J Med Genet. 2012 Nov;55(11):630-4. doi: 10.1016/j.ejmg.2012.07.005. Epub 2012 Aug 3.
Blepharophimosis-ptosis-epicanthus inversus syndrome (BPES) is a rare autosomal dominant disorder whose main features are the abnormal shape, position and alignment of the eyelids. Type I refers to BPES with female infertility from premature ovarian failure while type II is limited to the ocular features. A causative gene, FOXL2, has been localized to 3q23. We report a black female who carried a de novo chromosomal translocation and 3.13 Mb deletion at 3q23, 1.2 Mb 5' to FOXL2. This suggests the presence of distant cis regulatory elements at the extended FOXL2 locus. In spite of 21 protein coding genes in the 3.13 Mb deleted segment, the patient had no other malformation and a strictly normal psychomotor development at age 2.5 years. Our observation confirms panethnicity of BPES and adds to the knowledge of the complex cis regulation of human FOXL2 gene expression.
睑裂狭小-上睑下垂-内眦赘皮综合征(BPES)是一种罕见的常染色体显性疾病,其主要特征是眼睑的形状、位置和排列异常。I型指伴有卵巢早衰导致女性不孕的BPES,而II型仅限于眼部特征。一个致病基因FOXL2已定位到3q23。我们报告了一名黑人女性,她携带了一个新发的染色体易位以及3q23处3.13 Mb的缺失,该缺失位于FOXL2上游1.2 Mb的5'端。这表明在扩展的FOXL2基因座存在远距离顺式调控元件。尽管在3.13 Mb的缺失片段中有21个蛋白质编码基因,但该患者没有其他畸形,在2.5岁时精神运动发育完全正常。我们的观察证实了BPES的全种族性,并增加了对人类FOXL2基因表达复杂顺式调控的认识。