Petersen E M
S Afr Med J. 1986 Jan 4;69(1):63-8.
The clinical, radiological and biochemical findings in a black girl with a rare, inherited mucopolysaccharide storage disease, Sanfilippo's syndrome (mucopolysaccharidosis (MPS) III) type C, are described. Practical points concerning the biochemical diagnosis of this condition, arising from unusual characteristics of the deficient enzyme acetyl CoA: alpha-glucosaminide N-acetyltransferase, are discussed. Because phenotypic manifestations of mucopolysaccharidosis are mild in all four types of Sanfilippo's syndrome and screening tests for mucopolysacchariduria in these patients may be negative, many cases may be passed unrecognized or simply labelled as cases of nonspecific mental retardation. It is suggested that Sanfilippo's syndrome is grossly underdiagnosed in the RSA and clinicians are urged to develop a greater awareness of the existence, and often subtle presentation, of the condition.
本文描述了一名患有罕见遗传性粘多糖贮积病C型(Sanfilippo综合征,即粘多糖贮积症(MPS)III型)的黑人女孩的临床、放射学和生化检查结果。文中讨论了因缺乏的酶乙酰辅酶A:α-氨基葡萄糖苷N-乙酰转移酶的异常特性而产生的有关该病症生化诊断的实用要点。由于在所有四种类型的Sanfilippo综合征中粘多糖贮积症的表型表现都较为轻微,且这些患者的粘多糖尿筛查试验可能为阴性,许多病例可能未被识别或仅被简单标记为非特异性智力迟钝病例。有人认为,Sanfilippo综合征在南非共和国被严重漏诊,敦促临床医生提高对该病症的存在及其通常隐匿表现的认识。