• DOI:10.1177/088307388800300305
• Corpus ID: 38127378

Benign Paroxysmal Tonic Upgaze of Childhood

@article{Ouvrier1988BenignPT,
  title={Benign Paroxysmal Tonic Upgaze of Childhood},
  author={Robert A. Ouvrier and FrankA. Billson},
  journal={Journal of Child Neurology},
  year={1988},
  volume={3},
  pages={177 - 180}
}

• R. Ouvrier, F. Billson
• Published 1 July 1988
• Psychology, Medicine
• Journal of Child Neurology

Four cases of an apparently benign ocular motor syndrome of childhood are reported. The features of the disorder are: (1) onset in early life; (2) periods of constant or variably sustained tonic conjugate upward deviation of the eyes; (3) down-beating saccades in attempted downgaze, which are difficult to sustain below the neutral positions; (4) apparently normal horizontal eye movements; (5) frequent relief by sleep; (6) otherwise normal neurological findings apart from mild ataxia, chronic in one boy and at times of illness in one of the other patients; (7) absence of deterioration during observation spanning up to 15 years; (8) eventual improvement but with some residual ocular movement problems in two cases; (9) normal metabolic, electroencephalographic, and neuroradiologic investigations; (10) normal brain examination findings in one patient who died accidentally; and (11) an apparently good response to levodopa therapy in one patient. To the authors' knowledge, this condition has not been described previously. It may be a new levodopa-responsive condition, secondary to a localized neurotransmitter deficiency. (J Child Neurol 1988;3:177-180).