See Wing-Shan Q, So Chi-Chiu J, Cheuk Daniel Ka-Leung, van Wijk Richard, Ha Shau-Yin
Departments of Paediatrics & Adolescent Medicine.
Pathology, University of Hong Kong, Queen Mary Hospital, Hong Kong.
J Pediatr Hematol Oncol. 2020 Oct;42(7):e696-e697. doi: 10.1097/MPH.0000000000001582.
Glucose phosphate isomerase (GPI) deficiency is the second most common red blood cell enzymopathy involving the glycolysis pathway. It is an autosomal recessive disorder. Chronic hemolytic anemia is a common manifestation. The most severe one can present as hydrops fetalis. It can also be associated with neurologic dysfunction. We report a girl with severe hemolytic anemia at birth because of GPI deficiency. Enzyme activity assays were inconclusive because of previous blood transfusions. She was found to be compound heterozygous for 2 novel missense mutations, c.490C>A p.(Pro164Thr) and c.817C>T p.(Arg273Cys), in the GPI gene. Other than the chronic hemolytic anemia, she also has mild fine motor, gross motor delay, and developed cerebella ataxia since 5 years old.
葡萄糖磷酸异构酶(GPI)缺乏症是涉及糖酵解途径的第二常见红细胞酶病。它是一种常染色体隐性疾病。慢性溶血性贫血是常见表现。最严重的可表现为胎儿水肿。它还可能与神经功能障碍有关。我们报告一名因GPI缺乏症出生时即患有严重溶血性贫血的女孩。由于之前接受过输血,酶活性检测结果不明确。在GPI基因中,发现她为两种新型错义突变c.490C>A p.(Pro164Thr)和c.817C>T p.(Arg273Cys)的复合杂合子。除慢性溶血性贫血外,自5岁起她还存在轻度精细运动、大运动发育迟缓以及小脑共济失调。