DNA sequencing errors in molecular diagnostics of filamin myopathy

Clin Chem Lab Med. 2010 Oct;48(10):1409-14. doi: 10.1515/CCLM.2010.272. Epub 2010 Jun 27.

Abstract

Background: Filamin myopathy is a neuromuscular disorder manifesting with predominantly limb-girdle muscle weakness and in many patients with diaphragm paralysis and cardiomyopathy, caused by mutations in the filamin C (FLNC) gene. Molecular diagnosis of filamin myopathy based on direct DNA sequencing of coding exons is compromised by the presence of a high homology pseudogene (pseFLNC) located approximately 53.6 kb downstream of the functional FLNC gene on chromosome 7q.

Methods: Molecular cloning, RT-PCR and real-time PCR methods were used to detect sequence differences between the FLNC and pseFLNC that are implicated in known or potential molecular diagnostic errors. Overall, 50 patients with a phenotype resembling filamin myopathy have been screened for mutations in FLNC.

Results: FLNC sequence inconsistencies caused by the interference from pseFLNC were identified and diagnostic errors involving, in particular, the detection of the most frequent disease-causing FLNC p.W2710X mutation resolved. Mismatches between the FLNC and pseFLNC sequences were tabulated for future use.

Conclusions: We devise a strategy that allows one to discern mutations occurring in the functional FLNC from those harbored in pseFLNC, thus preventing possible complications in future research and patient genetic testing.

Publication types

  • Research Support, N.I.H., Intramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Base Sequence
  • Chromosomes, Human, Pair 7 / genetics
  • Cloning, Molecular
  • Contractile Proteins / genetics*
  • DNA / genetics*
  • Exons
  • Filamins
  • Humans
  • Microfilament Proteins / genetics*
  • Molecular Diagnostic Techniques*
  • Muscular Diseases / diagnosis*
  • Muscular Diseases / genetics*
  • Mutation
  • Reverse Transcriptase Polymerase Chain Reaction
  • Sequence Analysis, DNA*

Substances

  • Contractile Proteins
  • FLNC protein, human
  • Filamins
  • Microfilament Proteins
  • DNA