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[Paroxysmal tonic upgaze of childhood]
Mika Mochizuki1, Shin-ichiro Hamano, Sakiko Oshima
1Division of Neurology, Saitama Children's Medical Center, Iwatsuki, Saitama.
Insights
A child experienced paroxysmal tonic upgaze (PTU), a rare neurological condition causing abnormal eye movements. Symptoms resolved within 10 months without lasting impairment, indicating potential for spontaneous recovery.
Area of Science:
- Pediatric Neurology
- Ophthalmology
- Neurodevelopmental Disorders
Background:
- Paroxysmal tonic upgaze (PTU) is a rare neurological disorder characterized by intermittent upward deviations of the eyes.
- This case report details a child's experience with symptoms consistent with PTU following a febrile seizure.
Observation:
- The child presented with tonic upward eye deviation and downward saccades on attempted downward gaze after a febrile seizure.
- These abnormal eye movements occurred paroxysmally, resolved during sleep, and were unresponsive to L-dopa treatment.
Findings:
- The child's symptoms, including paroxysmal tonic upgaze and transient truncal ataxia, gradually subsided over 10 months.
- The patient achieved normal neurological development by 2 years and 6 months, with no residual impairment.
Implications:
- This case contributes to the understanding of paroxysmal tonic upgaze in pediatric neurology.
- The findings suggest that PTU can have a favorable prognosis with spontaneous resolution.
- Further research is needed to elucidate the underlying pathophysiology and long-term outcomes of PTU.
Abstract:
We described a child who developed paroxysmal abnormal eye movement. At the age of 12 months, she had a high fever and a febrile seizure. On the next day she showed tonic upward deviation of the eyes for 1 to 2 seconds, and downward saccades on attempted downward gaze. The upward deviation was repeated for a period of 2 to 3 hours, and disappeared during sleep. The administration of L-dopa was not effective. The symptoms subsided gradually over 10 months without other neurological impairment. She walked alone at 1 year and 6 months. At the beginning of her walking she showed truncal ataxia, but it gradually disappeared and her development was normal at the age of 2 years and 6 months. These abnormal eye movements and another symptoms were similar to paroxysmal tonic upgaze of childhood (PTU) that has been first described by Ouvrier and Billson (1988) as intermittent upward deviations of eyes. In Japan there was only one report of this syndrome with periventricular leukomalacia and hypomyelination.