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Published on: February 5, 2019
Long-term follow-up of benign positional vertical opsoclonus in infants: retrospective cohort
Amir Sternfeld1, Daniella Lobel2, Hana Leiba3
1Department of Ophthalmology, Rabin Medical Center-Beilinson Hospital, Petach Tikva, Israel.
Insights
Benign positional vertical opsoclonus in infants, also known as paroxysmal tonic downgaze, is a benign condition. This study found it resolves spontaneously with normal development, suggesting extensive work-ups may not be necessary.
Area of Science:
- Pediatric Ophthalmology
- Neuro-ophthalmology
- Infant Neurology
Background:
- Benign positional vertical opsoclonus (BPVO), or paroxysmal tonic downgaze, is an infant eye movement disorder.
- While often benign, it prompts extensive diagnostic work-ups.
- Sporadic cases suggest a benign course, necessitating further investigation.
Purpose of the Study:
- To describe the long-term follow-up of infants diagnosed with benign positional vertical opsoclonus.
- To evaluate the clinical course and outcomes of this condition.
- To determine if extensive investigations are warranted for BPVO.
Main Methods:
- Retrospective cohort study of infants diagnosed with rapid downgaze eye movements between 2012-2015.
- Clinical diagnosis by neuro-ophthalmologists based on specific eye movement findings.
- Data collection included demographics, symptoms, neurological/ophthalmological evaluations, imaging, and long-term outcomes.
Main Results:
- Six infants were included, presenting between several days and 12 weeks of age.
- Episodes were brief, frequent, and often occurred in the supine position.
- All infants showed gradual resolution of symptoms and achieved normal developmental milestones without pathological findings on extensive work-up.
Conclusions:
- Benign positional vertical opsoclonus may be more common than previously thought.
- The condition appears to affect otherwise healthy infants and resolves spontaneously.
- Given the favorable long-term outcome, a comprehensive clinical investigation may not be essential.
Background/Aims:
Benign positional vertical opsoclonus in infants, also described as paroxysmal tonic downgaze, is an unsettling phenomenon that leads to extensive work-up, although benign course has been reported in sporadic cases. We describe long-term follow-up of a series of infants with the phenomenon.
Methods:
This retrospective cohort included all infants diagnosed with rapid downgaze eye movement in 2012-2015 and followed until 2016. The databases of two medical centres were retrospectively reviewed. Benign positional vertical opsoclonus was diagnosed based on clinical findings of experienced neuro-ophthalmologists. Data were collected on demographics, symptoms and signs, neuro-ophthalmological and neurological evaluations, and outcome. Imaging studies were reviewed. Main outcome measures were long-term outcome and findings of the thorough investigation.
Results:
The cohort included six infants. All infants were born at term. Age at presentation was several days to 12 weeks. Episodes lasted a few seconds and varied in frequency from <10 to dozens per day. In five infants, symptoms occurred in the supine position. There was a wide variability in the work-up without any pathological findings. Follow-up ranged from 1 to 2.5 years. Ocular symptoms gradually decreased until resolution. Infants reached normal developmental milestones.
Conclusions:
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.
