Baclofen for patients with congenital periodic alternating nystagmus.

To describe the clinical effect of baclofen on a group of patients with congenital periodic alternating nystagmus.
A retrospective review of case notes was carried out of all patients with congenital periodic alternating nystagmus (PAN) treated with baclofen between 1999 and 2004. Eight patients were identified, 6 males and 2 females with a mean age of 21 years (range 9 to 34 years). Clinical data were recorded for all patients pre- and post-treatment with the GABA agonist baclofen. Adverse effects of the treatment were recorded and a questionnaire was constructed to evaluate patient satisfaction with the treatment.
All 8 patients had an abnormal head posture (AHP) before treatment which improved following treatment in 4 patients, one of whom had recurrence following treatment withdrawal. Binocular Snellen visual acuity (VA) improved by one line in 4 cases, while none of the other 4 patients suffered any loss of vision from the treatment. Three of the eight patients have continued on treatment long-term, in one case for 6 years. In the other five, treatment was withdrawn due to side effects in 4 cases, and in the fifth due to a lack of effect. The most commonly reported side effect was drowsiness, which occurred in 3 patients. Using a patient-centered survey, complete responses were obtained from 6 of the 8 patients treated. Five patients were either pleased or very pleased that they had tried the treatment. The reasons given were: improved vision or head posture, an appreciation of slowing of ocular movements, improved cosmesis and improved confidence.
Baclofen may be effective in a select group of patients with congenital PAN and a trial of treatment may be worthwhile, prior to considering surgical intervention in this condition.
AuthorsR M Comer, E L M Dawson, J P Lee
JournalStrabismus (Strabismus) Vol. 14 Issue 4 Pg. 205-9 (Dec 2006) ISSN: 0927-3972 [Print] England
PMID17162442 (Publication Type: Journal Article)
Chemical References
  • GABA Agonists
  • Baclofen
  • Adolescent
  • Adult
  • Baclofen (adverse effects, therapeutic use)
  • Child
  • Female
  • GABA Agonists (adverse effects, therapeutic use)
  • Humans
  • Male
  • Nystagmus, Congenital (drug therapy)
  • Patient Satisfaction
  • Retrospective Studies
  • Vision, Binocular

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