Nelson J, Kenny B, O'Hara D, Harper A, Broadhead D
Department of Medical Genetics, Northern Ireland.
J Clin Pathol. 1993 Apr;46(4):370-1. doi: 10.1136/jcp.46.4.370.
Mucopolysaccharidosis type VII (MPS VII, beta glucuronidase deficiency) has been described in association with non-immune hydrops fetalis. Three consecutive pregnancies in an itinerant family, which resulted in stillbirths caused by non-immune hydrops are described. The parents were closely related and there was a strong family history of storage disorders. The main clue to the diagnosis, however, came from the presence of pronounced foamy cytoplasmic change in the villous Hofbauer cells of the placenta. This raised the possibility of an inherited metabolic storage disorder. The parents were subsequently shown to have beta glucuronidase activities in the heterozygous range in leucocytes and fibroblasts which suggested that the non-immune hydrops was caused by beta glucuronidase deficiency.
VII型粘多糖贮积症(MPS VII,β-葡萄糖醛酸酶缺乏症)已被描述与非免疫性胎儿水肿有关。本文描述了一个流动家庭连续三次怀孕,均因非免疫性胎儿水肿导致死产。父母近亲结婚,且有很强的遗传性贮积病家族史。然而,诊断的主要线索来自胎盘绒毛霍夫鲍尔细胞中明显的泡沫状细胞质变化。这增加了遗传性代谢贮积病的可能性。随后发现父母白细胞和成纤维细胞中的β-葡萄糖醛酸酶活性处于杂合子范围,这表明非免疫性胎儿水肿是由β-葡萄糖醛酸酶缺乏引起的。