Pazzaglia U E, Beluffi G, Danesino C, Frediani P V, Pagani G, Zatti G
Clinica Ortopedica dell'Università di Pavia, Italy.
Pediatr Radiol. 1989;20(1-2):80-4. doi: 10.1007/BF02010640.
Evolution of the early bone lesions in two children with mucolipidosis 2 was followed from birth. The progression of the bone changes did not differ from healing of rickets. Low levels of 1,25-(OH)2-D3 were found in one child and he was treated with vitamin D; resolution of the rachitic changes was more rapid than in the untreated child. It is suggested that in mucolipidosis 2 bone reacts to two independent factors, one controlling calcium metabolism, the other depending on the primary lysosomal enzyme defect. Since ricket-like features are not present in the other mucolipidoses or mucopolysaccharidoses, the defect of calcium metabolism seems to be related to the specific enzyme defect of mucolipidosis 2.
对两名患有黏脂贮积症Ⅱ型的儿童从出生起就追踪其早期骨病变的演变过程。骨变化的进展与佝偻病的愈合情况并无差异。在一名儿童中发现1,25 -(OH)₂-D₃水平较低,对其进行了维生素D治疗;与未治疗的儿童相比,佝偻病性变化的消退更为迅速。有人提出,在黏脂贮积症Ⅱ型中,骨骼对两个独立因素产生反应,一个控制钙代谢,另一个取决于原发性溶酶体酶缺陷。由于其他黏脂贮积症或黏多糖贮积症中不存在类似佝偻病的特征,钙代谢缺陷似乎与黏脂贮积症Ⅱ型的特定酶缺陷有关。