Literature DB >> 8127074

Carbonic anhydrase II deficiency in three unrelated Japanese patients.

S Aramaki1, I Yoshida, M Yoshino, M Kondo, Y Sato, K Noda, R Jo, A Okue, N Sai, F Yamashita.   

Abstract

Three Japanese patients with carbonic anhydrase II (CAII) deficiency from three families were described. The parents of one patient were unrelated, the parents of each of the other two patients were first cousins. All the patients had renal tubular acidosis, osteopetrosis, symmetrical cerebral calcification and mental retardation. They exhibited poor activity and poor appetite in the neonatal period, and then developed psychomotor retardation. Two of them were diagnosed as having osteopetrosis at 10 months and 36 years of age, respectively, and the other as having osteomalacia at 28 years of age. All patients had recurrent episodes of muscle weakness. The CAII enzyme activity and protein levels in red blood cells in each of the three patients were deficient. Their parents exhibited approximately 50% normal levels of CAII activity and protein. This is the first report of patients with CAII deficiency in the Japanese population.

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Year:  1993        PMID: 8127074     DOI: 10.1007/bf00711514

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  15 in total

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Journal:  J Pharmacol Exp Ther       Date:  1960-09       Impact factor: 4.030

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4.  [Osteopetrosis and renal tubular acidosis. 2 cases of this association in a sibship].

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Journal:  Arch Fr Pediatr       Date:  1972-03

Review 5.  Biochemical genetics of carbonic anhydrase.

Authors:  R E Tashian; N D Carter
Journal:  Adv Hum Genet       Date:  1976

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Authors:  R E Tashian; D Hewett-Emmett; S J Dodgson; R E Forster; W S Sly
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8.  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

9.  Carbonic anhydrase II deficiency syndrome in a Belgian family is caused by a point mutation at an invariant histidine residue (107 His----Tyr): complete structure of the normal human CA II gene.

Authors:  P J Venta; R J Welty; T M Johnson; W S Sly; R E Tashian
Journal:  Am J Hum Genet       Date:  1991-11       Impact factor: 11.025

10.  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

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