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Related Experiment Videos

Best's macular dystrophy with a macular hole.

M Mehta1, O Katsumi, S Tetsuka

  • 1Department of Ophthalmology, Eye Research Institute of Retina Foundation, Boston.

Acta Ophthalmologica
|February 1, 1991
PubMed
Summary

Best

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Area of Science:

  • Ophthalmology
  • Genetics
  • Retinal Diseases

Background:

  • Best's dystrophy is a rare inherited retinal disorder.
  • It is characterized by the accumulation of lipofuscin-like material in the retinal pigment epithelium.
  • Macular holes are a common complication of various retinal diseases.

Observation:

  • This study reports two cases of Best's dystrophy.
  • In these cases, typical Best's dystrophy was observed in one eye, while a macular hole was present in the contralateral eye.
  • Pattern Reversal Visual Evoked Potential (PVER) testing was performed in one patient.

Findings:

  • The co-occurrence of macular holes and Best's dystrophy is extremely rare, with only one previous case reported.
  • PVER results indicated significantly subnormal responses in both eyes, suggesting widespread macular impairment.
  • This impairment was present even at the vitelliform stage of Best's dystrophy.

Implications:

  • The findings suggest that significant functional impairment can occur during the early, vitelliform stage of Best's dystrophy.
  • The progression from the vitelliform stage to the development of a macular hole remains unclear.
  • Further research is needed to understand the underlying mechanisms and clinical course of this rare condition.

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