Baumann R J, Markesbery W R
Ann Neurol. 1978 Dec;4(6):531-6. doi: 10.1002/ana.410040609.
Lymphocytes from 3 children with a form of juvenile amaurotic idiocy (characterized by retinal blindness, progressive dementia, and extrapyramidal motor disturbance) were studied by electron microscopy. Numerous fingerprint profiles (FP) were found in vacuolated lymphocytes from all 3 patients and an asymptomatic younger sibling of 1 patient who subsequently became symptomatic. We propose that the combination of this clinical picture and vacuolated lymphocytes with FP is sufficiently distinctive for clinical and research purposes until the biochemical defect is discovered. Wider utilization of ultrastructural study of lymphocytes should increase the number of children diagnosed and allow detection of asymptomatic patients.
对3名患有某种青少年黑蒙性白痴(特征为视网膜失明、进行性痴呆和锥体外系运动障碍)的儿童的淋巴细胞进行了电子显微镜研究。在所有3名患者以及1名患者的无症状弟弟(该弟弟后来出现症状)的空泡化淋巴细胞中发现了大量指纹图谱(FP)。我们认为,在发现生化缺陷之前,这种临床症状与带有FP的空泡化淋巴细胞的组合对于临床和研究目的而言具有足够的独特性。更广泛地利用淋巴细胞的超微结构研究应能增加被诊断儿童的数量,并能检测出无症状患者。