Back to Search
Start Over
Two novel CAII mutations causing carbonic anhydrase II deficiency syndrome in two unrelated Chinese families.
- Source :
-
Metabolic brain disease [Metab Brain Dis] 2015 Aug; Vol. 30 (4), pp. 989-97. Date of Electronic Publication: 2015 Feb 27. - Publication Year :
- 2015
-
Abstract
- The carbonic anhydrase II (CAII) deficiency syndrome is a rare autosomal recessive osteopetrosis with renal tubular acidosis (RTA) and cerebral calcifications (MIM259730). CAII deficiency syndrome is caused by mutations in the gene CAII, which encodes the enzyme carbonic anhydrase II. CAII mutations are rarely reported in the Asian population. Here, we described two unrelated CAII deficiency families of Chinese Han origin with clinical and genetic analysis. Altogether, 106 subjects, including 2 probands, 4 unaffected family members from two non-consanguineous Chinese families, and 100 healthy controls were recruited. All seven exons and the exon-intron boundaries of the CAII gene were amplified and directly sequenced. Reverse transcription PCR (RT-PCR) was used to study the effect of splice site mutation. All clinical and biochemical parameters of the probands were collected. Two novel mutations of CAII gene were identified by mutational analysis: A nonsense mutation in exon 4 (c.T381C p.Y127X) in both families; a splice mutation at the splice donor site of intron 3 (c.350+2T>C, IVS3+2T>C) in one family. The splice-site mutation causes exon 3 skipping in patient's mRNA resulting in an in-frame deletion and a novel premature stop codon. These mutations were predicted to result in a loss of function of CAII. This is the first report of CAII deficiency syndrome in Chinese population. Our findings extent the spectrum of CAII mutations observed in patients with CAII deficiency syndrome.
- Subjects :
- Adolescent
Carbonic Anhydrase II chemistry
Carbonic Anhydrases genetics
Child, Preschool
Female
Humans
Male
Pedigree
Protein Structure, Secondary
Acidosis, Renal Tubular diagnosis
Acidosis, Renal Tubular genetics
Asian People genetics
Carbonic Anhydrase II genetics
Carbonic Anhydrases deficiency
Mutation genetics
Osteopetrosis diagnosis
Osteopetrosis genetics
Urea Cycle Disorders, Inborn diagnosis
Urea Cycle Disorders, Inborn genetics
Subjects
Details
- Language :
- English
- ISSN :
- 1573-7365
- Volume :
- 30
- Issue :
- 4
- Database :
- MEDLINE
- Journal :
- Metabolic brain disease
- Publication Type :
- Academic Journal
- Accession number :
- 25720518
- Full Text :
- https://doi.org/10.1007/s11011-015-9660-6