Konstantinidou A E, Anninos H, Dertinger S, Nonni A, Petersen M, Karadimas C, Havaki S, Marinos E, Akman H O, DiMauro S, Patsouris E
Department of Pathology, Medical Faculty, National Kapodistrian University of Athens, Greece.
Placenta. 2008 Apr;29(4):378-81. doi: 10.1016/j.placenta.2008.01.005. Epub 2008 Mar 4.
Glycogen storage disease type IV (GSD IV) is a rare autosomal recessive disorder caused by glycogen branching enzyme (GBE) deficiency and resulting in the storage of abnormal glycogen (polyglucosan). Prenatal diagnosis is based on biochemical assay of GBE activity or on mutation analysis, but polyglucosan can also be identified histologically in fetal tissues. We document placental involvement at 25 and 35 weeks of gestation in two cases with genetically confirmed GSD IV. Intracellular inclusions were seen mainly in the extravillous trophoblast. Our findings suggest the possibility of prenatal diagnosis by histological evaluation of placental biopsies.
IV型糖原贮积病(GSD IV)是一种罕见的常染色体隐性疾病,由糖原分支酶(GBE)缺乏引起,导致异常糖原(多聚葡萄糖)蓄积。产前诊断基于GBE活性的生化检测或突变分析,但多聚葡萄糖也可在胎儿组织中通过组织学方法鉴定。我们记录了两例经基因确诊的GSD IV患者在妊娠25周和35周时胎盘受累的情况。细胞内包涵体主要见于绒毛外滋养层。我们的研究结果提示,通过胎盘活检的组织学评估进行产前诊断具有可能性。