Bennett M J, Galloway J H, Cartwright I J, Gillis W S, Hosking G P
Department of Chemical Pathology, Sheffield Children's Hospital, U.K.
Neuropediatrics. 1990 Nov;21(4):202-5. doi: 10.1055/s-2008-1071496.
Total cellular and phospholipid fatty acids were analyzed in erythrocytes and platelets from six patients with juvenile neuronal ceroid-lipofuscinosis (Spielmeyer-Vogt, Batten disease, JNCL). The results were compared to those of age-matched controls. The amounts of total fatty acid and the phospholipid classes, phosphatidylcholine (PC), phosphatidylethanolamine (PE), and phosphatidylserine (PS) were significantly decreased in patients when related to cellular protein. The reductions in total fatty acids were 27% in erythrocytes and 50% in platelets. Erythrocyte PC reduction was 36%, PE was 44% and PS 27%. There were no major qualitative differences in the phospholipid fatty acids, suggesting that the fatty acid composition of the reduced phospholipid was normal, and that there is a generalized phospholipid deficiency in these cells in JNCL. This was not related to nutritional status. The pathogenesis of Batten disease may be related to abnormal membrane function resulting from this marked phospholipid deficiency.
对6例青少年神经元蜡样脂褐质沉积症(施皮尔曼 - 沃格特病、巴顿病、JNCL)患者的红细胞和血小板中的总细胞脂肪酸和磷脂脂肪酸进行了分析。将结果与年龄匹配的对照组进行比较。与细胞蛋白相关时,患者的总脂肪酸以及磷脂类,即磷脂酰胆碱(PC)、磷脂酰乙醇胺(PE)和磷脂酰丝氨酸(PS)的含量显著降低。红细胞中总脂肪酸减少27%,血小板中减少50%。红细胞中PC减少36%,PE减少44%,PS减少27%。磷脂脂肪酸没有重大的质的差异,这表明减少的磷脂的脂肪酸组成是正常的,并且在JNCL的这些细胞中存在普遍的磷脂缺乏。这与营养状况无关。巴顿病的发病机制可能与这种明显的磷脂缺乏导致的膜功能异常有关。