Chitayat D, Nakagawa S, Marion R W, Sachs G S, Hahm S Y, Goldman H S
Department of Pediatrics, Albert Einstein College of Medicine, Bronx, New York 10467.
Am J Med Genet. 1988 Nov;31(3):527-32. doi: 10.1002/ajmg.1320310307.
We report on 3 Puerto Rican brothers with the clinical and laboratory findings of aspartylglucosaminuria (AGU). Their parents were first cousins. The affected sibs have the "cardinal" manifestations of AGU, including developmental disabilities, progressive "coarsening" of the face, and early onset of hepatosplenomegaly. Biochemical studies showed elevated levels of urinary aspartylglucosamine and very low activity of aspartylglucosaminidase(AGA) in cultured fibroblasts. With long term follow-up, previously undescribed manifestations were noted, including radiographic evidence of spondylolysis and spondylolisthesis in early childhood and development of macro-orchidism during puberty. This family shows that AGU is not limited to individuals of Finnish background, but that the gene is panethnic in distribution and that additional changes, not previously noted, may present with advancing age.
我们报告了3名患有天冬氨酰葡糖胺尿症(AGU)临床及实验室检查结果的波多黎各兄弟。他们的父母是近亲。受影响的同胞具有AGU的“主要”表现,包括发育障碍、面部逐渐“变粗”以及肝脾肿大早发。生化研究显示尿中天冬氨酰葡糖胺水平升高,培养的成纤维细胞中天冬氨酰葡糖胺酶(AGA)活性极低。经过长期随访,发现了以前未描述的表现,包括幼儿期椎骨溶解和脊椎滑脱的影像学证据以及青春期巨睾症的发展。这个家系表明AGU并不局限于芬兰血统的个体,而是该基因在各民族中均有分布,并且随着年龄增长可能会出现以前未注意到的其他变化。