Rapid diagnosis of Wilson disease by a 28-mutation panel: real-time amplification refractory mutation system in diagnosing acute Wilsonian liver failure

Clin Chim Acta. 2008 Dec;398(1-2):39-42. doi: 10.1016/j.cca.2008.08.002. Epub 2008 Aug 8.

Abstract

Background: Wilson disease is one of the commonest inherited and potentially fatal yet treatable liver disorders. About 5-27% patients present with acute liver failure and require prompt chelation therapy and life-saving liver transplantation. Diagnosis during acute liver failure is particularly difficult with short time allowance. Direct molecular diagnosis remains the most decisive tool but is often hindered by demanding techniques and numerous mutations. We developed a one-step, 3-h, reproducible, and accurate real-time amplification refractory mutation system which can simultaneously detect 28 ATP7B mutations.

Methods: Primers were designed to complement the mutant sequence at the 3' end. The mutations were p.S105X, p.Q511X, p.R616Q, p.S693P, p.S693C, p.R778L, p.A874V, p.T888P, p.R919G, p.T935M, p.P992L, p.M1025R, p.D1047V, p.I1148T, p.R1156H, p.T1178A, p.V1216M, p.P1273Q, p.G1281C, p.R1320S, p.V1334D, p.V176SfsX28, p.G869EfsX4, IVS3+1G>T, IVS4-1G>C, IVS4-5T>G, IVS6+9A>G, and IVS9+5G>T. Reaction was performed using QuantiTect SYBR Green PCR Master Mix on an Applied Biosystems StepOne thermal cycler. Values of the threshold cycle were compared between normal and mutant alleles.

Results: Primers of all mutations were highly specific with absence of wild-type amplification. All the results were validated by direct DNA sequencing.

Conclusions: This rapid and cost-efficient method allows wide mutation coverage, rendering the SYBR-green assay feasible and attractive for large-scale routine application.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Alleles
  • Benzothiazoles
  • Cost-Benefit Analysis
  • DNA / genetics
  • DNA Mutational Analysis / economics
  • DNA Mutational Analysis / methods*
  • DNA Primers
  • Diamines
  • Female
  • Fluorescent Dyes
  • Hepatolenticular Degeneration / complications
  • Hepatolenticular Degeneration / diagnosis*
  • Hepatolenticular Degeneration / genetics*
  • Humans
  • Liver Failure, Acute / diagnosis*
  • Liver Failure, Acute / etiology
  • Liver Failure, Acute / genetics*
  • Mutation / genetics
  • Organic Chemicals
  • Quinolines
  • Reverse Transcriptase Polymerase Chain Reaction

Substances

  • Benzothiazoles
  • DNA Primers
  • Diamines
  • Fluorescent Dyes
  • Organic Chemicals
  • Quinolines
  • SYBR Green I
  • DNA