Literature DB >> 11399210

Acute intermittent porphyria: novel missense mutations in the human hydroxymethylbilane synthase gene.

R B Ramdall1, L Cunha, K H Astrin, D R Katz, K E Anderson, M Glucksman, S S Bottomley, R J Desnick.   

Abstract

PURPOSE: To identify mutations in families with acute intermittent porphyria, an autosomal dominant inborn error of metabolism that results from the half-normal activity of the third enzyme in the heme biosynthetic pathway, hydroxymethylbilane synthase.
METHODS: Mutations were identified by direct solid phase sequencing.
RESULTS: Two novel missense mutations E80G and T78P and three previously reported mutations, R173W, G111R, and the splice site lesion, IVS1+1, were detected, each in an unrelated proband. The causality of the novel missense mutations was demonstrated by expression studies.
CONCLUSION: These findings provide for the precise diagnosis of carriers in these families and further expand the molecular heterogeneity of AIP.

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Year:  2000        PMID: 11399210     DOI: 10.1097/00125817-200009000-00004

Source DB:  PubMed          Journal:  Genet Med        ISSN: 1098-3600            Impact factor:   8.822


  2 in total

1.  Mutation hotspots in the human porphobilinogen deaminase gene: recurrent mutations G111R and R173Q occurring at CpG motifs.

Authors:  X Schneider-Yin; M Hergersberg; M M Schuurmans; A Gregor; E I Minder
Journal:  J Inherit Metab Dis       Date:  2004       Impact factor: 4.982

Review 2.  Acute Intermittent Porphyria: An Overview of Therapy Developments and Future Perspectives Focusing on Stabilisation of HMBS and Proteostasis Regulators.

Authors:  Helene J Bustad; Juha P Kallio; Marta Vorland; Valeria Fiorentino; Sverre Sandberg; Caroline Schmitt; Aasne K Aarsand; Aurora Martinez
Journal:  Int J Mol Sci       Date:  2021-01-12       Impact factor: 5.923

  2 in total

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