Terespolsky D, Clarke J T, Blaser S I
Department of Pediatrics and Genetics, Hospital for Sick Children, Toronto, Ontario, Canada.
J Inherit Metab Dis. 1996;19(6):775-81. doi: 10.1007/BF01799172.
We report on clinical and neuroradiological findings in two patients with fucosidosis type II; a 7-year-old Jordanian boy and a 3 1/2-year-old Anglo-Canadian girl. This rare, autosomal recessive disorder is caused by deficiency of lysosomal alpha-fucosidase and is manifested clinically by progressive mental and motor deterioration, coarse facies, growth retardation, recurrent infections, dysostosis multiplex, angiokeratoma corporis diffusum, visceromegaly and seizures. Cranial CT and magnetic resonance imaging showed density and signal abnormalities in the thalamus, globus pallidus and internal capsules bilaterally, as well as progressive CT density alterations in supratentorial white matter including the internal medullary laminae of the thalami and the internal capsules.
我们报告了两名II型岩藻糖苷贮积症患者的临床和神经放射学检查结果;一名7岁的约旦男孩和一名3岁半的英裔加拿大女孩。这种罕见的常染色体隐性疾病是由溶酶体α-岩藻糖苷酶缺乏引起的,临床上表现为进行性智力和运动功能衰退、面容粗糙、生长发育迟缓、反复感染、多发性骨发育异常、全身性弥漫性血管角质瘤、内脏肿大和癫痫发作。头颅CT和磁共振成像显示双侧丘脑、苍白球和内囊有密度和信号异常,以及幕上白质包括丘脑内髓板和内囊的CT密度逐渐改变。