Fukuda T, Sugie H, Sugie Y, Ito M, Tsurui S, Igarashi Y
Department of Pediatrics, Hamamatsu University School of Medicine, Shizuoka.
No To Hattatsu. 1994 Nov;26(6):493-7.
Muscle, erythrocyte and/or leukocyte phosphorylase b kinase (PBK) activities were measured in 5 patients and in 2 families. The relation between phenotype and enzyme deficiency in clinically variable samples based on measurement of erythrocytes, leukocytes or muscle was described. Three patients showed the enzyme deficiency in liver and the disease was transmitted as an X-linked recessive trait. In these cases, our results suggested that carrier detection was more reliable with erythrocytes than leukocytes. Two patients showed enzyme deficiency in muscle, and the mode of inheritance was not clear. In these cases, muscle biopsies were necessary for diagnosis.
对5名患者和2个家族的肌肉、红细胞和/或白细胞磷酸化酶b激酶(PBK)活性进行了测量。基于红细胞、白细胞或肌肉测量结果,描述了临床症状多样的样本中表型与酶缺乏之间的关系。3名患者肝脏中出现酶缺乏,疾病呈X连锁隐性遗传。在这些病例中,我们的结果表明,通过红细胞检测携带者比通过白细胞检测更可靠。2名患者肌肉中出现酶缺乏,遗传模式不明确。在这些病例中,肌肉活检对于诊断是必要的。