A novel FOXF1 mutation associated with alveolar capillary dysplasia and coexisting colobomas and hemihyperplasia

被引:3
|
作者
Geddes, G. C. [1 ,2 ,3 ]
Dimmock, D. P. [1 ,2 ,3 ]
Hehir, D. A. [1 ,2 ]
Helbling, D. C. [3 ]
Kirkpatrick, E. [1 ,2 ]
Loomba, R. [1 ,2 ]
Southern, J. [4 ]
Waknitz, M. [3 ]
Scharer, G. [1 ,2 ,3 ]
Konduri, G. G. [1 ,2 ]
机构
[1] Med Coll Wisconsin, Childrens Hosp Wisconsin, Dept Pediat, Milwaukee, WI 53226 USA
[2] Med Coll Wisconsin, Childrens Hosp Wisconsin, Childrens Res Inst, Milwaukee, WI 53226 USA
[3] Med Coll Wisconsin, Human & Mol Genet Ctr, Milwaukee, WI 53226 USA
[4] Med Coll Wisconsin, Dept Pathol, Milwaukee, WI 53226 USA
关键词
MISALIGNED PULMONARY VEINS;
D O I
10.1038/jp.2014.187
中图分类号
R71 [妇产科学];
学科分类号
100211 ;
摘要
Alveolar capillary dysplasia (ACD) is a rare and lethal cause of hypoxic respiratory failure in the neonate. Here we describe a term neonate with ACD that was found with a previously unreported p.Arg86Pro mutation in the FOXF1 (Forkhead Box-F1) gene and coexisting congenital anomalies, including colobomas of the iris and hemihyperplasia. This unique clinical presentation may indicate a novel, yet unconfirmed disease association for mutations in the FOXF1 gene. Rapid mutation analysis in FOXF1 may provide noninvasive early confirmation of ACD in neonates with respiratory failure and can aid in clinical decision making.
引用
收藏
页码:155 / 157
页数:3
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