Albini A, Pontz B, Mensing H, Cantz M, Müller P K
Padiatr Padol. 1983;18(3):263-7.
Certain clinical symptoms such as hernias or joint contracture in patients with mucopolysaccharidoses (MPS) cannot be explained as direct consequences of the disturbed glycosaminoglycan metabolism. They may be related to secondary changes of connective tissue components. The glycoprotein fibronectin is a constituent of connective tissue with a high affinity to polyanions such as heparan sulfate or heparin. Fibronectin in addition is a potent stimulus for fibroblasts to migrate chemotactically. We studied this cell property in MPS fibroblasts. The chemotactic activity of all MPS types was diminished. MPS II fibroblasts were chemotactically inactive. When the cells were corrected for the lacking enzyme by adding conditioned medium from control fibroblasts, the chemotactic migration increased except for MPS type II cells. The known enzyme defect in the degradation process of glycosaminoglycans in MPS results in lysosomal storage of degradation products and in addition causes changes of other cell properties.
黏多糖贮积症(MPS)患者出现的某些临床症状,如疝气或关节挛缩,不能解释为糖胺聚糖代谢紊乱的直接后果。它们可能与结缔组织成分的继发性变化有关。糖蛋白纤连蛋白是结缔组织的一种成分,对诸如硫酸乙酰肝素或肝素等多阴离子具有高亲和力。此外,纤连蛋白是成纤维细胞进行趋化性迁移的有效刺激物。我们在MPS成纤维细胞中研究了这种细胞特性。所有类型的MPS的趋化活性均降低。MPS II型成纤维细胞无趋化活性。当通过添加对照成纤维细胞的条件培养基来纠正细胞中缺乏的酶时,除MPS II型细胞外,趋化性迁移增加。MPS中糖胺聚糖降解过程中已知的酶缺陷导致降解产物在溶酶体中蓄积,此外还会引起其他细胞特性的改变。