LRP2

Clinical Characteristics

Most individuals with DBS, which is always caused by biallelic LRP2 mutations, have these characteristics:

Craniofacial features

  • macrocephaly at birth
  • broad forehead and frontal bossing
  • hypertelorism
  • prominent brows
  • downward palpebral slant
  • flat nasal bridge
  • short nose with indented tip
  • low-set and posterior angulated ears

Eyes and vision

  • enlarged globes
  • progression to high grade myopia (-20 diopters bilaterally)
  • significantly increased risk of retinal detachment
  • iris hypoplasia and coloboma
  • subcapsular opacities and cataracts

Ears and hearing

  • progression to bilateral moderate or profound sensorineural hearing loss (SNHL)

Central nervous system

  • agenesis of the corpus callosum (ACC)

Kidney

  • low-molecular-weight proteinuria (LMWP)
  • reduced tubular uptake of LRP2 ligands

Other structural congenital anomalies

  • congenital diaphragmatic hernia (CDH)
  • omphalocele

Less commonly reported findings are:

Eyes

  • leukocoria and retinal dystrophy
  • exotropia
  • vitreous anomalies

Kidney

  • nephrocalcinosis and nephrolithiasis
  • microalbuminuria
  • progression to renal dysfunction

Brain/Central nervous system

  • developmental delay
  • seizures

Other structural congenital anomalies

  • umbilical hernia
  • wide metopic suture
  • congenital heart disease (e.g. ventricular septal defect, double-outlet right ventricle)
  • urogenital anomalies