Genetic and electrophysiological characteristics of recurrent acute pancreatitis

J Pediatr Gastroenterol Nutr. 2015 May;60(5):675-9. doi: 10.1097/MPG.0000000000000623.

Abstract

Objectives: The aim was to present the workup of patients with acute recurrent pancreatitis (ARP) for genetic analysis and electrophysiological testing.

Methods: Patients with ARP with unknown etiology were referred for genetic testing and evaluation of cystic fibrosis transmembrane conductor regulator (CFTR) function by nasal potential difference (NPD) testing.

Results: A total of 67 patients were evaluated. The mean age was 23 ± 17 years (median 17.0 years, range 1.5-72 years); 90% were Jewish and 10% Arab. Ten (15%) patients carried PRSS1 gene mutation (K23R(7), R122H(2), and D21A(1)). One patient had K172E/- (chymotrypsin C [CTRC]) mutation, 1 had I42M (serine protease inhibitor Kazal type 1 [SPINK1])/V235I (CTRC) together with ΔF508/5T, 1 patient had R67H (SPINK1)/V235I (CTRC), and 1 patient had V235I (CTRC)/-. Ten of 67 (15%) patients submitted for CFTR gene testing carried mutations (ΔF508/L997F, ΔF508/5T(11TG), W1282/5T(12TG), W1282X/Y1014C, ΔF508/R31C, R117H/-, R117H/Y1014C, D1152H/-, 5T(11TG)/-, and L997F/-). Fifty-four (80%) patients underwent sweat testing. Of these, 5 had sweat chloride ≥60 mEq/L, and 22 patients had sweat chloride from 40 to 60 mEq/L. Of the 56 (83%) patients had nasal potential difference testing, 4 (6%) with abnormal results.

Conclusions: One-third (34%) of patients with ARP carry mutations for hereditary pancreatitis including rare mutations (K23R), and 12.5% have evidence of cftr mutations and 10% had CFTR dysfunction underscoring the importance of genetic and functional workup of these patients.

MeSH terms

  • Acute Disease
  • Adolescent
  • Adult
  • Aged
  • Arabs / genetics
  • Carrier Proteins / genetics
  • Child
  • Child, Preschool
  • Chlorides / analysis
  • Chymotrypsin / genetics
  • Cystic Fibrosis Transmembrane Conductance Regulator / genetics
  • Cystic Fibrosis Transmembrane Conductance Regulator / metabolism
  • Electrophysiological Phenomena
  • Female
  • Humans
  • Infant
  • Israel
  • Jews / genetics
  • Male
  • Membrane Potentials
  • Middle Aged
  • Nose / physiopathology*
  • Pancreatitis / ethnology
  • Pancreatitis / genetics*
  • Pancreatitis / physiopathology*
  • Recurrence
  • Respiratory Mucosa / physiopathology*
  • Sweat / chemistry
  • Trypsin / genetics
  • Trypsin Inhibitor, Kazal Pancreatic
  • Young Adult

Substances

  • Carrier Proteins
  • Chlorides
  • SPINK1 protein, human
  • Cystic Fibrosis Transmembrane Conductance Regulator
  • Trypsin Inhibitor, Kazal Pancreatic
  • Chymotrypsin
  • chymotrypsin C
  • PRSS1 protein, human
  • Trypsin