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Identification of a novel mutation in an Indian patient with CAII deficiency syndrome.
- Source :
-
Journal of postgraduate medicine [J Postgrad Med] 2010 Oct-Dec; Vol. 56 (4), pp. 290-2. - Publication Year :
- 2010
-
Abstract
- 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.
- Subjects :
- Acidosis, Renal Tubular diagnosis
Acidosis, Renal Tubular enzymology
Adult
Calcinosis diagnosis
Calcinosis enzymology
Calcinosis genetics
Humans
Intellectual Disability diagnosis
Intellectual Disability enzymology
Male
Osteopetrosis diagnosis
Osteopetrosis enzymology
Pedigree
Phenotype
Sequence Analysis, DNA
Syndrome
Acidosis, Renal Tubular genetics
Carbonic Anhydrase II deficiency
Carbonic Anhydrase II genetics
Frameshift Mutation genetics
Intellectual Disability genetics
Osteopetrosis genetics
Subjects
Details
- Language :
- English
- ISSN :
- 0972-2823
- Volume :
- 56
- Issue :
- 4
- Database :
- MEDLINE
- Journal :
- Journal of postgraduate medicine
- Publication Type :
- Academic Journal
- Accession number :
- 20935402
- Full Text :
- https://doi.org/10.4103/0022-3859.70944