FAM111B Mutation Is Associated With Inherited Exocrine Pancreatic Dysfunction

Pancreas. 2016 Jul;45(6):858-62. doi: 10.1097/MPA.0000000000000529.

Abstract

Objectives: Few genetic causes of exocrine pancreatic dysfunction have been described to date. We identified a family with multiple affected members manifesting exocrine pancreatic dysfunction. Additional associated features included facial rash, sparse hair, hypohidrosis, and swelling of the extremities. The transmission pattern of these clinical features was consistent with an autosomal dominant mode of inheritance. The 2 proband siblings also had transient elevated liver transaminases with hepatic steatosis early in life. This study identifies the genetic cause of exocrine pancreatic dysfunction in this family.

Methods: Whole exome sequencing was performed to identify the genetic cause of exocrine pancreatic dysfunction.

Results: A heterozygous germline in-frame deletion in the gene FAM111B (c.1261_1263delAAG, p.Lys421del) cosegregated with the phenotype: the variant was present in all affected relatives genotyped and absent in all unaffected relatives genotyped. The variant is also absent from public control sequence databases.

Conclusions: Our findings implicate FAM111B in autosomal dominantly inheritable exocrine pancreatic dysfunction.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Adult
  • Aged
  • Amino Acid Sequence
  • Base Sequence
  • Blotting, Western
  • Cell Cycle Proteins / genetics*
  • Cell Cycle Proteins / metabolism
  • Child
  • Exome / genetics
  • Family Health
  • Female
  • Genetic Predisposition to Disease / genetics*
  • Germ-Line Mutation*
  • Humans
  • Male
  • Middle Aged
  • Pancreas, Exocrine / metabolism*
  • Pancreas, Exocrine / physiopathology
  • Pancreatic Diseases / genetics*
  • Pancreatic Diseases / metabolism
  • Pedigree
  • Phenotype
  • Sequence Analysis, DNA / methods
  • Sequence Homology, Nucleic Acid
  • Siblings

Substances

  • Cell Cycle Proteins
  • FAM111B protein, human