N34S mutation in the SPINK1 gene is not associated with alternative splicing

Pancreas. 2007 May;34(4):423-8. doi: 10.1097/mpa.0b013e3180335fd0.

Abstract

Objectives: Previous studies have shown an association between chronic pancreatitis (CP) and mutations, especially the N34S mutation, in the serine protease inhibitor Kazal type 1 (SPINK1) gene. But the underlying molecular mechanisms are unknown. The aberrant splicing caused by the cosegregating intronic mutations might play a role, but this hypothesis has not been tested. We here examined the messenger RNA sequences of the SPINK1 gene in patients carrying the mutations.

Methods: RNA was isolated from the surgically resected pancreas of 2 CP patients carrying the homozygous N34S mutation and from the gastric biopsy specimen of a CP patient carrying the heterozygous [-215G>A; IVS3+2T>C] mutation. The entire coding region of the SPINK1 gene was amplified by reverse transcription-polymerase chain reaction, subcloned, and sequenced. The level of the wild-type SPINK1 transcript was assessed by real-time polymerase chain reaction.

Results: Alternative splicing was not associated with the N34S mutation. On the other hand, the [-215G>A; IVS3+2T>C] mutation caused skipping of whole exon 3, where the trypsin binding site is located. This mutated protein was predicted to consist of 63 amino acids: deletion of amino acid sequence from residues 30 to 64 and shifting of reading frame at amino acid 65 with a novel stop codon. The expression of the wild-type SPINK1 transcript was decreased to 62% of the healthy control in the CP patient carrying the heterozygous [-215G>A; IVS3+2T>C] mutation.

Conclusions: Splicing mutation might represent a mechanism for SPINK1-associated CP, but the N34S mutation is not associated with alternative splicing.

MeSH terms

  • Adolescent
  • Adult
  • Alternative Splicing*
  • Amino Acid Sequence
  • Asparagine
  • Base Sequence
  • Carrier Proteins / chemistry
  • Carrier Proteins / genetics*
  • Carrier Proteins / metabolism
  • Cloning, Molecular
  • DNA Mutational Analysis
  • Exons
  • Female
  • Heterozygote
  • Homozygote
  • Humans
  • Introns
  • Male
  • Middle Aged
  • Molecular Sequence Data
  • Mutation*
  • Pancreatitis, Chronic / genetics*
  • Pancreatitis, Chronic / metabolism
  • Promoter Regions, Genetic
  • RNA, Messenger / metabolism
  • Reverse Transcriptase Polymerase Chain Reaction
  • Sequence Analysis, Protein
  • Serine
  • Trypsin Inhibitor, Kazal Pancreatic

Substances

  • Carrier Proteins
  • RNA, Messenger
  • SPINK1 protein, human
  • Serine
  • Trypsin Inhibitor, Kazal Pancreatic
  • Asparagine