Shivaprasad C, Paliwal P, Khadgawat R, Sharma A
Department of Anatomy, All India Institute of Medical Sciences, New Delhi, India.
J Postgrad Med. 2010 Oct-Dec;56(4):290-2. doi: 10.4103/0022-3859.70944.
Carbonic anhydrase II (CAII) deficiency syndrome characterized by osteopetrosis (OP), renal tubular acidosis (RTA), and cerebral calcifications is caused by mutations in the carbonic anhydrase 2 (CA2) gene. Severity of this disorder varies depending on the nature of the mutation and its effect on the protein. We present here, the clinical and radiographic details along with, results of mutational analysis of the CA2 gene in an individual clinically diagnosed with renal tubular acidosis, osteopetrosis and mental retardation and his family members to establish genotype-phenotype correlation. A novel homozygous deletion mutation c.251delT was seen in the patient resulting in a frameshift and a premature stop codon at amino acid position 90 generating a truncated protein leading to a complete loss of function and a consequential deficiency of the enzyme making this a pathogenic mutation. Confirmation of clinical diagnosis by molecular methods is essential as the clinical features of the CAII deficiency syndrome are similar to other forms of OP but the treatment modalities are different. Genetic confirmation of the diagnosis at an early age leads to the timely institution of therapy improving the growth potential, reduces other complications like fractures, and aids in providing prenatal testing and genetic counseling to the parents planning a pregnancy.
碳酸酐酶II(CAII)缺乏综合征的特征为骨质石化(OP)、肾小管酸中毒(RTA)和脑钙化,由碳酸酐酶2(CA2)基因突变引起。这种疾病的严重程度因突变的性质及其对蛋白质的影响而异。我们在此呈现一名临床诊断为肾小管酸中毒、骨质石化和智力发育迟缓的个体及其家庭成员的临床和影像学详细信息,以及CA2基因的突变分析结果,以建立基因型与表型的相关性。在该患者中发现了一种新的纯合缺失突变c.251delT,导致移码并在第90位氨基酸处产生一个过早的终止密码子,产生截短的蛋白质,导致功能完全丧失以及该酶的相应缺乏,使其成为一种致病突变。通过分子方法确认临床诊断至关重要,因为CAII缺乏综合征的临床特征与其他形式的骨质石化相似,但治疗方式不同。早期通过基因确诊可及时进行治疗,改善生长潜力,减少骨折等其他并发症,并有助于为计划怀孕的父母提供产前检测和遗传咨询。