Long-term follow-up of benign positional vertical opsoclonus in infants: retrospective cohort

Abstract

Our identification of six patients in only 3 years suggests benign positional vertical opsoclonus may be more prevalent than previously described. In our experience, it affects otherwise healthy infants and resolves spontaneously. In view of the good long-term outcome, a comprehensive clinical investigation may not be necessary.

A Case of Rapidly Resolving Abnormal Vertical Eye Movements in a Late‐Preterm Neonate with Germinal Matrix Hemorrhages

“…They have been previously related to benign opsoclonus and PTD cases, 3,6 favoring a possible pathophysiological parallel between these eye movement changes. Some authors also suggest these eye movements can reflect an immature myelination of the vertical eye movement systems 1 and a mild (even if non-specific) myelination delay was found in our patient. However, the rapid resolution of the paroxysmal eye movements does not fully support both theories.…”

“…Neonates may present with several involuntary eye movements, such as transient neonatal opsoclonus (TNO), nystagmus, seizures and paroxysmal tonic upgaze (PTU) and downgaze (PTD). 1,2 Even if often transient and benign, PTU can be accompanied by neurological deficits (namely ataxia and developmental delay), that can persist overtime, and be secondary to upper brainstem structural lesions or genetic channelopathies (eg, CACNA1A mutations). 2 PTD is commonly regarded as a more benign entity, provided hydrocephalus has been excluded, 1,2 but TNO, long recognized in preterm infants, 2,3 should also motivate the exclusion of structural causes.…”

“…1,2 Even if often transient and benign, PTU can be accompanied by neurological deficits (namely ataxia and developmental delay), that can persist overtime, and be secondary to upper brainstem structural lesions or genetic channelopathies (eg, CACNA1A mutations). 2 PTD is commonly regarded as a more benign entity, provided hydrocephalus has been excluded, 1,2 but TNO, long recognized in preterm infants, 2,3 should also motivate the exclusion of structural causes. Lately, a vertical variant, with a positional preference (positional vertical opsoclonus, PVO), has also been described in twins, suggesting a possible genetic role.…”