Department of Pediatrics, Konkuk University Hospital, Konkuk University School of Medicine, Korea.
Gene. 2012 Oct 10;507(2):170-3. doi: 10.1016/j.gene.2012.06.090. Epub 2012 Jul 5.
Gaucher's disease is caused by a deficiency of glucocerebrosidase (GBA) and results in the accumulation of glucocerebroside within macrophages. We report on a 33(+2) gestational week premature infant whose family history was significant for a previously undiagnosed premature sibling with similar clinical features, including severe hydrops fetalis, hepatosplenomegaly, skin lesions at birth followed by death. The diagnosis of Gaucher's disease type 2 in the present case was based on postmortem pathological findings and a subsequent gene analysis that indicated a heterozygous condition for the novel deletion mutation at GBA cDNA nucleotide position 630 resulting in the frameshift (Pro171fsX21) in exon 6 and a G→A transition mutation at GBA cDNA nucleotide position 887 (Arg257Gln) in exon 7.
戈谢病是由于葡萄糖脑苷脂酶(GBA)缺乏引起的,导致葡萄糖脑苷脂在巨噬细胞内堆积。我们报告了一例 33(+2)孕周的早产儿,其家族史中有一个先前未诊断的早产同胞,具有相似的临床特征,包括严重的胎儿水肿、肝脾肿大、出生时皮肤损伤,随后死亡。本病例戈谢病 2 型的诊断基于尸检病理发现和随后的基因分析,表明 GBA cDNA 核苷酸位置 630 处的新型缺失突变导致移码(Pro171fsX21),并在外显子 6 中存在 G→A 转换突变,GBA cDNA 核苷酸位置 887(Arg257Gln)在外显子 7 中。