Pendular nystagmus in patients with peroxisomal assembly disorder

Arch Neurol. 1998 Apr;55(4):554-8. doi: 10.1001/archneur.55.4.554.

Abstract

Background: Pendular nystagmus commonly occurs in congenital and acquired disorders of myelin.

Objective: To characterize the nystagmus in 3 siblings with an infantile form of an autosomal recessive peroxisomal assembly disorder causing leukodystrophy.

Design: We examined visual function and measured eye movements using infrared oculography. We noted changes in eye speed and frequency before and after the administration of gabapentin to 1 patient.

Results: All 3 siblings showed optic atrophy and pendular nystagmus that was predominantly horizontal, at a frequency of 3 to 6 Hz, with phase shifts of 45 degrees to 80 degrees between the oscillations of each eye. Gabapentin administered to 1 child caused a modest improvement of vision and the reduction of the velocity and frequency of oscillations in the eye with worse nystagmus.

Conclusion: The pendular nystagmus in these patients was due to their leukodystrophy and may have a similar pathogenesis to the oscillations seen in other disorders affecting central myelin.

Publication types

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

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Eye Movements / physiology
  • Female
  • Genes, Recessive*
  • Humans
  • Male
  • Nystagmus, Pathologic / genetics*
  • Optic Atrophy / genetics
  • Peroxisomal Disorders / genetics
  • Peroxisomal Disorders / physiopathology*
  • Vision Screening