Bull's-eye maculopathy in an infant with Leigh disease

Am J Ophthalmol. 2006 Jul;142(1):186-7. doi: 10.1016/j.ajo.2006.02.051.

Abstract

Purpose: To report a bull's-eye maculopathy-like fundus abnormality in an infant with Leigh disease.

Design: Observational case report.

Methods: We reviewed the medical, ophthalmic, and genetic records of an eight-month-old boy who presented with hypotonia and bilaterally decreased vision.

Results: The ophthalmic examination revealed poor fixation, marked hyperopia, attenuated retinal vessels, and bull's-eye maculopathy. A white blood cell mitochondrial DNA analysis demonstrated a T to G mutation at position 8993 in the mitochondrial ATPase 6 gene. A skeletal muscle biopsy was homoplasmic for this mutation, consistent with a severe mitochondrial disorder.

Conclusion: Leigh disease should be included in the differential diagnosis of patients presenting with neurologic deficits and a bull's-eye maculopathy.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adenosine Triphosphatases / genetics
  • Atrophy
  • Brain / pathology
  • DNA, Mitochondrial / analysis
  • Diagnosis, Differential
  • Humans
  • Infant
  • Leigh Disease / complications*
  • Leigh Disease / diagnosis
  • Macula Lutea / pathology*
  • Magnetic Resonance Imaging
  • Male
  • Mitochondria, Muscle / enzymology
  • Mitochondria, Muscle / genetics
  • Muscle Hypotonia / diagnosis
  • Mutation
  • Retinal Diseases / diagnosis
  • Retinal Diseases / etiology*

Substances

  • DNA, Mitochondrial
  • Adenosine Triphosphatases