Bifid nose as the sole manifestation of BNAR syndrome, a FREM1-related condition

Clin Genet. 2020 Nov;98(5):515-516. doi: 10.1111/cge.13821. Epub 2020 Sep 14.

Abstract

BNAR syndrome (MIM608980) is a very rare condition: nine cases belonging to three unrelated families were reported since its first description in 2002. The distinctive clinical feature is the bifidity of the tip of the nose and its association with anorectal and/or renal anomalies. Its molecular basis consisting of biallelic FREM1 missense or nonsense mutations was elucidated after studying the original Egyptian family and was confirmed in two families originating from Afghanistan and Pakistan. We describe a fourth family originating from Turkey with signs challenging the diagnostic criteria suggested by the description of the three reported families.

Publication types

  • Case Reports
  • Letter

MeSH terms

  • Abnormalities, Multiple / genetics*
  • Abnormalities, Multiple / physiopathology
  • Coloboma / genetics
  • Coloboma / physiopathology
  • Egypt / epidemiology
  • Humans
  • Hypertelorism / genetics*
  • Hypertelorism / physiopathology
  • Male
  • Musculoskeletal Abnormalities / genetics
  • Musculoskeletal Abnormalities / physiopathology
  • Nose / abnormalities*
  • Nose / physiopathology
  • Nose Diseases / genetics*
  • Nose Diseases / physiopathology
  • Pakistan / epidemiology
  • Phenotype
  • Receptors, Interleukin / genetics*
  • Respiratory System Abnormalities
  • Turkey / epidemiology

Substances

  • Frem1 protein, human
  • Receptors, Interleukin

Supplementary concepts

  • Bifid nose