A lysosomal storage disease induced by Ipomoea carnea in goats in Mozambique
A lysosomal storage disease induced by Ipomoea carnea in goats in Mozambique
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DOI：
10.1177/104063879901100310
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发表时间：
1999-05-01
影响因子：
1.5
通讯作者：
Welman, WG
中科院分区：
文献类型：
--
作者：
de Balogh, KKIM;Dimande, AP;Welman, WG
在莫桑比克一个村庄的山羊中观察到一种新的植物诱导的溶酶体贮积病。受影响的动物为共济失调，伴有头部震颤和眼球震颤。由于缺乏合适的饲料，这些动物食用了一种生长在村里的外来树篱植物，这种植物被鉴定为Ipomoea carnea（灌木状牵牛花，旋花科）。通过饲喂I. carnea植物材料给山羊。在急性病例中，脑和脊髓的组织学变化包括与轴突球体形成相关的神经元和胶质细胞的广泛胞质空泡化。超微结构上，神经元胞质贮液泡呈膜结合，与溶酶体一致。在急性病例中，还在小肠粘膜下和肠系膜丛的神经元、肾小管上皮细胞以及脾脏和淋巴结的巨噬细胞吞噬细胞中发现细胞质空泡化。在慢性病例中，大脑的残留改变主要表现为小脑病变，其特征在于浦肯野神经元的丢失和浦肯野细胞层的胶质增生。分析I。通过气相色谱-质谱法对鼠尾草植物材料的分析确定了甘露糖苷酶抑制剂苦马豆素和2种糖苷酶抑制剂，花蒿碱B-2和花蒿碱C-1的存在，这与山羊中植物诱导的α-甘露糖苷中毒一致。所描述的胆积症类似于由摄入疯草（黄芪属和棘豆属）和毒豌豆（苦豆属）诱导的溶酶体胆积症。
A novel plant-induced lysosomal storage disease was observed in goats from a village in Mozambique. Affected animals were ataxic, with head tremors and nystagmus. Because of a lack of suitable feed, the animals consumed an exotic hedge plant growing in the village that was identified as Ipomoea carnea (shrubby morning glory, Convolvulaceae). The toxicosis was reproduced by feeding I. carnea plant material to goats. In acute cases, histologic changes in the brain and spinal cord comprised widespread cytoplasmic vacuolation of neurons and glial cells in association with axonal spheroid formation. Ultrastructurally, cytoplasmic storage vacuoles in neurons were membrane bound and consistent with lysosomes. Cytoplasmic vacuolation was also found in neurons in the submucosal and mesenteric plexuses in the small intestine, in renal tubular epithelial cells, and in macrophage-phagocytic cells in the spleen and lymph nodes in acute cases. Residual alterations in the brain in chronic cases revealed predominantly cerebellar lesions characterized by loss of Purkinje neurons and gliosis of the Purkinje cell layer. Analysis of I. carnea plant material by gas chromatography-mass spectrometry established the presence of the mannosidase inhibitor swainsonine and 2 glycosidase inhibitors, calystegine B-2 and calystegine C-1, consistent with a plant-induced a-mannosidosis in the goats. The described storage disorder is analogous to the lysosomal storage diseases induced by ingestion of locoweeds (Astragalus and Oxytropis) and poison peas (Swainsona).