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OTX2

OTX2

Orthodenticle homeobox 2; a transcription factor critical for retinal photoreceptor development and bipolar cell differentiation. De novo heterozygous mutations can cause early-onset retinal dystrophy/LCA. Also associated with anophthalmia, microphthalmia, and pituitary dysfunction.

No Targeted Treatment Yet

Chromosomal Locus

14q22.3

Inheritance

Autosomal Dominant

Frequency

<1% of LCA cases

Key Clinical Features

  • 1No official LCA subtype number assigned
  • 2Autosomal dominant — typically de novo mutations
  • 3Rare cause of LCA/early-onset retinal dystrophy
  • 4Electronegative ERG suggesting photoreceptor and bipolar cell dysfunction
  • 5Can be associated with pituitary dysfunction

Syndromic Associations

Patients with OTX2 mutations should be monitored for potential involvement of other organs:

  • Microphthalmia syndromic 5 (MCOPS5)
  • Pituitary hormone deficiency