Klingensmith W C, Eikman E A, Maumenee I, Wanger H N
J Nucl Med. 1975 Nov;16(11):1002-6.
Mucopolysaccharidoses (MPS) are inherited disorders of lysosomal enzymes. We have examined the sites of accumulation of intravenously injected 99mTc-sulfur colloid in order to assess the regional distribution of phagocytic function in ten patients with MPS: three with Type VI (Maroteaux-Lamy). Increased lung uptake was observed in 22 of 40 studies (55%) on the five patients with MPZ Type II but in none of the 38 studies on patients with other MPS types. All MPS patients had diffuse recticuloendothelial (RE) marrow hypoplasia, despite normal or nearly normal hematocrits and hemoglobin levels, suggestion dissociation of the phagocytic and erythropoietic elements of the marrow. The eight patients with MPZ Types I and II all had hepatomegaly and increased splenic uptake. Seven of these patients with MPS Types III and VI did not have hepatosplenomegaly. These studies indicate that the lysosoma enzymic defect of MPS results in widespread abnormalities of the distribution of phagocytic function in the liver, spleen, bone marrow, and probably the lung as well.
黏多糖贮积症(MPS)是溶酶体酶的遗传性疾病。我们检测了静脉注射的99mTc-硫胶体的蓄积部位,以评估10例MPS患者吞噬功能的区域分布:3例为VI型(马罗-拉米氏病)。在5例II型MPS患者的40项研究中有22项(55%)观察到肺部摄取增加,但在其他MPS类型患者的38项研究中均未观察到。所有MPS患者均有弥漫性网状内皮(RE)骨髓发育不全,尽管血细胞比容和血红蛋白水平正常或接近正常,提示骨髓吞噬和造血成分解离。8例I型和II型MPS患者均有肝肿大和脾脏摄取增加。其中7例III型和VI型MPS患者没有肝脾肿大。这些研究表明,MPS的溶酶体酶缺陷导致肝脏、脾脏、骨髓以及可能还有肺部吞噬功能分布的广泛异常。