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1例伴有FGFR3 P.R248C突变且存活超过新生儿期的Ⅰ型致死性骨发育不良病例

A THANATOPHORIC DYSPLASIA TYPE I CASE WITH A FGFR3 P.R248C MUTATION AND SURVIVAL BEYOND THE NEONATAL PERIOD.

作者信息

Sahin S, Ograg H, Aslan E Atas, Akcan A B, Turkmen M Kaynak, Moosa S, Elcioglu N H

出版信息

Genet Couns. 2016;27(4):513-517.

Abstract

A Thanatophoric dysplasia, is a severe congenital anomaly which mostly causes stillbirth or death of the affected baby within hours due to respiratory insufficiency. The diagnosis of TD is typically suspected on ultrasound during the second trimester of pregnancy, when severe shortening of the long bones, frontal bossing, flattened vertebrae, and short ribs that result in a narrow thorax and bell-shaped abdomen, can be seen. Here, we present a case with prenatal ultrasonographic findings suggestive of TD, and highlight the patient's postnatal dysmorphic features and typical radiographic findings. The definitive diagnosis of TD type I (TDI) was made postnatally, when molecular genetic analysis revealed the previously described p.R248C mutation in FGFR3. This case is reported due to its relative long life span and the definitive molecular diagnosis that could be made during hospitalization.

摘要

致死性发育不良是一种严重的先天性异常,大多会导致受影响的婴儿因呼吸功能不全在数小时内死产或死亡。在妊娠中期进行超声检查时通常会怀疑患有致死性发育不良,此时可看到长骨严重缩短、前额突出、椎体扁平以及肋骨短小,从而导致胸廓狭窄和腹部呈钟形。在此,我们报告一例产前超声检查结果提示致死性发育不良的病例,并着重介绍患者出生后的畸形特征和典型的影像学表现。出生后通过分子遗传学分析发现FGFR3基因存在先前描述的p.R248C突变,从而确诊为I型致死性发育不良(TDI)。报告该病例是因其相对较长的存活时间以及住院期间能够做出明确的分子诊断。

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