Corrocher R, Guadagnin L, de Gironcoli M, Girelli D, Guarini P, Olivieri O, Caffi S, Stanzial A M, Ferrari S, Grigolini L
Istituto di Patologia Medica, Università di Verona, Italy.
J Endocrinol Invest. 1989 Jul-Aug;12(7):475-81. doi: 10.1007/BF03350737.
The fatty acid composition of erythrocyte membrane, the glutathione-peroxidase activity of erythrocytes and platelets, the production of malondialdehyde by platelets and the activity of the main systems of transmembrane cation transport have been studied in 5 members of a family, 2 of whom affected by Laurence-Moon-Barter-Biedl Syndrome. A remarkable increase of polyunsaturated fatty acids (particularly arachidonic acid) and of cholesterol/phospholipid molar ratio has been noted. This pattern of membrane lipids was associated to an increment of malondialdehyde production and an increase activity of glutathione-peroxidase. Serum retinol and a-tocopherol were in the normal range, whereas serum selenium was low in 3 out of 5 members. Moreover, the alteration of membrane lipids was associated to a decrease of the maximal velocity of Li-Na countertransport. We speculate that the enrichment of polyunsaturated fatty acids on the cell membranes may represent a condition favoring the lipoperoxidation and therefore the development of the retinitis pigmentosa characteristic feature of Laurence-Moon-Barter-Biedl Syndrome.
对一个家族的5名成员进行了研究,其中2人患有劳伦斯-穆恩-巴特尔-比德尔综合征,检测了他们红细胞膜的脂肪酸组成、红细胞和血小板的谷胱甘肽过氧化物酶活性、血小板丙二醛的生成以及跨膜阳离子转运主要系统的活性。结果发现多不饱和脂肪酸(尤其是花生四烯酸)以及胆固醇/磷脂摩尔比显著增加。这种膜脂质模式与丙二醛生成增加和谷胱甘肽过氧化物酶活性增强有关。血清视黄醇和α-生育酚在正常范围内,而5名成员中有3人的血清硒含量较低。此外,膜脂质的改变与锂-钠逆向转运的最大速度降低有关。我们推测细胞膜上多不饱和脂肪酸的富集可能是一种有利于脂质过氧化的情况,从而导致劳伦斯-穆恩-巴特尔-比德尔综合征特征性的视网膜色素变性的发展。