Literature DB >> 25720518

Two novel CAII mutations causing carbonic anhydrase II deficiency syndrome in two unrelated Chinese families.

Qianqian Pang1, Xuan Qi, Yan Jiang, Ou Wang, Mei Li, Xiaoping Xing, Jin Dong, Weibo Xia.   

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.

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Year:  2015        PMID: 25720518     DOI: 10.1007/s11011-015-9660-6

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.584


  26 in total

1.  Seven novel mutations in carbonic anhydrase II deficiency syndrome identified by SSCP and direct sequencing analysis.

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Journal:  Hum Mutat       Date:  1997       Impact factor: 4.878

2.  Albers-Schönberg disease (autosomal dominant osteopetrosis, type II) results from mutations in the ClCN7 chloride channel gene.

Authors:  E Cleiren; O Bénichou; E Van Hul; J Gram; J Bollerslev; F R Singer; K Beaverson; A Aledo; M P Whyte; T Yoneyama; M C deVernejoul; W Van Hul
Journal:  Hum Mol Genet       Date:  2001-12-01       Impact factor: 6.150

3.  A new heterozygous mutation (R714C) of the osteopetrosis gene, pleckstrin homolog domain containing family M (with run domain) member 1 (PLEKHM1), impairs vesicular acidification and increases TRACP secretion in osteoclasts.

Authors:  Andrea Del Fattore; Rachele Fornari; Liesbeth Van Wesenbeeck; Fenna de Freitas; Jean-Pierre Timmermans; Barbara Peruzzi; Alfredo Cappariello; Nadia Rucci; Giovanni Spera; Miep H Helfrich; Wim Van Hul; Silvia Migliaccio; Anna Teti
Journal:  J Bone Miner Res       Date:  2008-03       Impact factor: 6.741

4.  Molecular basis of human carbonic anhydrase II deficiency.

Authors:  D E Roth; P J Venta; R E Tashian; W S Sly
Journal:  Proc Natl Acad Sci U S A       Date:  1992-03-01       Impact factor: 11.205

5.  SNX10 mutations define a subgroup of human autosomal recessive osteopetrosis with variable clinical severity.

Authors:  Alessandra Pangrazio; Anders Fasth; Andrea Sbardellati; Paul J Orchard; Kimberly A Kasow; Jamal Raza; Canan Albayrak; Davut Albayrak; Olivier M Vanakker; Barbara De Moerloose; Ashok Vellodi; Luigi D Notarangelo; Claire Schlack; Gabriele Strauss; Jörn-Sven Kühl; Elena Caldana; Nadia Lo Iacono; Lucia Susani; Uwe Kornak; Ansgar Schulz; Paolo Vezzoni; Anna Villa; Cristina Sobacchi
Journal:  J Bone Miner Res       Date:  2013-05       Impact factor: 6.741

6.  Refined structure of human carbonic anhydrase II at 2.0 A resolution.

Authors:  A E Eriksson; T A Jones; A Liljas
Journal:  Proteins       Date:  1988

7.  Structure of native and apo carbonic anhydrase II and structure of some of its anion-ligand complexes.

Authors:  K Håkansson; M Carlsson; L A Svensson; A Liljas
Journal:  J Mol Biol       Date:  1992-10-20       Impact factor: 5.469

8.  Carbonic anhydrase II deficiency identified as the primary defect in the autosomal recessive syndrome of osteopetrosis with renal tubular acidosis and cerebral calcification.

Authors:  W S Sly; D Hewett-Emmett; M P Whyte; Y S Yu; R E Tashian
Journal:  Proc Natl Acad Sci U S A       Date:  1983-05       Impact factor: 11.205

9.  Carbonic anhydrase II deficiency a novel mutation.

Authors:  Sheela Nampoothiri; Yair Anikster
Journal:  Indian Pediatr       Date:  2009-06       Impact factor: 1.411

10.  Involvement of PLEKHM1 in osteoclastic vesicular transport and osteopetrosis in incisors absent rats and humans.

Authors:  Liesbeth Van Wesenbeeck; Paul R Odgren; Fraser P Coxon; Annalisa Frattini; Pierre Moens; Bram Perdu; Carole A MacKay; Els Van Hul; Jean-Pierre Timmermans; Filip Vanhoenacker; Ruben Jacobs; Barbara Peruzzi; Anna Teti; Miep H Helfrich; Michael J Rogers; Anna Villa; Wim Van Hul
Journal:  J Clin Invest       Date:  2007-04       Impact factor: 14.808

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  4 in total

1.  Molecular dynamics study of human carbonic anhydrase II in complex with Zn(2+) and acetazolamide on the basis of all-atom force field simulations.

Authors:  Thierry O Wambo; Liao Y Chen; Stanton F McHardy; Andrew T Tsin
Journal:  Biophys Chem       Date:  2016-05-18       Impact factor: 2.352

2.  Report of Another Mutation Proven Case of Carbonic Anhydrase II Deficiency.

Authors:  Amit Kumar Satapathy; Swati Pandey; Madhumita Roy Chaudhary; Arvind Bagga; Madhulika Kabra; Kornak Uwe; Neerja Gupta
Journal:  J Pediatr Genet       Date:  2018-11-18

Review 3.  Genetics of Osteopetrosis.

Authors:  Eleonora Palagano; Ciro Menale; Cristina Sobacchi; Anna Villa
Journal:  Curr Osteoporos Rep       Date:  2018-02       Impact factor: 5.096

4.  Carbonic Anhydrase II Deficiency: A Rare Case of Severe Obstructive Sleep Apnea.

Authors:  Emanuela di Palmo; Marcella Gallucci; Elena Tronconi; Rosalba Bergamaschi; Salvatore Cazzato; Claudio La Scola; Giampaolo Ricci; Andrea Pession
Journal:  Front Pediatr       Date:  2018-07-31       Impact factor: 3.418

  4 in total

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