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1 型辛普森-高柏-比姆尔综合征,出生参数正常。

Simpson-Golabi-Behmel syndrome type 1 with normal birth parameters.

机构信息

Paediatrics & Adolescent Medicine, The University of Hong Kong Li Ka Shing Faculty of Medicine, Hong Kong, Hong Kong

Hong Kong Children's Hospital, Hong Kong, Hong Kong.

出版信息

BMJ Case Rep. 2024 Mar 4;17(3):e247864. doi: 10.1136/bcr-2021-247864.

Abstract

A newborn baby born at 34 weeks and 5 days gestation was admitted for prematurity, dysmorphic features and congenital heart defects. Antenatal scan at 21 weeks showed a large-for-gestational-age foetus with a large abdominal circumference and liver, ventricular septal defect, right prominent renal pelvis and echogenic bowel. Antenatal genetic tests for overgrowth syndromes were negative. The mother had early onset pre-eclampsia. After birth, an overgrowth syndrome was still suspected despite the baby having normal birth parameters. Raw data of the trio whole exome sequencing from the amniocentesis sample were manually inspected. Hemizygous exon 7 deletion in the GPC3 gene was found, and a postnatal diagnosis of Simpson-Golabi-Behmel syndrome, a rare overgrowth syndrome, was made. This case report discusses the significance of antenatal findings, an atypical presentation of a rare syndrome and the obstacles of diagnostic genetic testing.

摘要

一名孕 34 周+5 天的新生儿因早产、畸形特征和先天性心脏缺陷入院。21 周的产前扫描显示胎儿为大于胎龄儿,腹部和肝脏较大,室间隔缺损,右肾盂突出,肠回声增强。针对过度生长综合征的产前基因检测结果为阴性。母亲患有早发性子痫前期。出生后,尽管婴儿的出生参数正常,但仍怀疑存在过度生长综合征。对羊水穿刺样本的三人体外显子组测序原始数据进行了手动检查。发现 GPC3 基因第 7 外显子杂合缺失,产后诊断为辛普森-戈拉比-贝姆尔综合征,这是一种罕见的过度生长综合征。本病例报告讨论了产前发现、罕见综合征的非典型表现以及诊断性基因检测的障碍的意义。

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