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SMAD4 基因杂合致病性变异导致的 Myhre 综合征的产前诊断表现为颈项透明层增厚和心脏异常。

Prenatal diagnosis of Myhre syndrome with a heterozygous pathogenic variant in SMAD4 gene presented with thick nuchal translucency and cardiac abnormalities.

机构信息

Department of Obstetrics and Gynaecology, Queen Mary Hospital, Hong Kong, Hong Kong.

Department of Health, Clinical Genetic Service, Hong Kong, Hong Kong.

出版信息

Prenat Diagn. 2023 Sep;43(10):1366-1369. doi: 10.1002/pd.6414. Epub 2023 Aug 2.

Abstract

Prenatal testing was performed in a 39-year-old Chinese pregnant woman referred for increased nuchal translucency measuring 5.7 mm. Non-invasive prenatal testing and SNP array study on amniotic fluid samples were normal. Whole exome sequencing (WES) was initiated further as the fetus had pericardial effusion of 1.2 mm, thickened myocardium over the right ventricular lateral wall and aberrant right subclavian artery. A detailed fetal echocardiogram also revealed persistent left superior vena cava and dilated coronary sinus at 20 weeks. From whole exome sequencing of the trio, a de novo heterozygous variant NM_005359.5(SMAD4): c.1499T>C (p.Ile500Thr) was detected. This pathogenic variant has been reported in the postnatal case cohort of Myhre syndrome. This condition is characterized by facial dysmorphism, intellectual disability, hearing loss, skeletal abnormalities and potential life threatening respiratory or cardiovascular manifestations. Termination of pregnancy was performed at 23 weeks. Small chins, pre-axial polydactyly, brachydactyly and clinodactyly were noted in the abortus. Ultrasound findings of increased nuchal translucency, thickened myocardium and pericardial effusion prompted further genetic evaluation for the prenatal diagnosis of Myhre syndrome by whole exome sequencing.

摘要

一位 39 岁的中国孕妇因颈项透明层增厚 5.7mm 而接受了产前检查。对羊水样本进行了无创性产前检查和 SNP 微阵列研究,结果均正常。由于胎儿有心包积液 1.2mm、右心室外侧壁心肌增厚和右锁骨下动脉异常,进一步进行了全外显子组测序(WES)。详细的胎儿超声心动图还显示 20 周时持续存在左上腔静脉和扩张的冠状窦。通过对三个人的全外显子组测序,发现了一个新的杂合性变异 NM_005359.5(SMAD4): c.1499T>C (p.Ile500Thr)。该致病性变异已在 Myhre 综合征的新生儿病例队列中报道。该病症的特征是面部畸形、智力障碍、听力损失、骨骼异常和潜在威胁生命的呼吸或心血管表现。在 23 周时终止了妊娠。流产儿有小下巴、前轴多指畸形、短指畸形和指内弯。颈项透明层增厚、心肌增厚和心包积液的超声发现提示通过全外显子组测序进行产前诊断 Myhre 综合征的进一步遗传评估。

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