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Atypical oculopalatal tremor as the presentation of vertebral artery dolichoectasia

Authors Vanikieti K, Cheecharoen P, Jindahra P, Lueangaram S, Padungkiatsagul T

Received 27 March 2016

Accepted for publication 18 June 2016

Published 6 September 2016 Volume 2016:9 Pages 273—277

DOI https://doi.org/10.2147/IMCRJ.S109357

Checked for plagiarism Yes

Review by Single-blind

Peer reviewers approved by Dr Colin Mak

Peer reviewer comments 2

Editor who approved publication: Professor Ronald Prineas


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Kavin Vanikieti,1 Piyaphon Cheecharoen,2 Panitha Jindahra,3 Sirin Lueangaram,4 Tanyatuth Padungkiatsagul1

1
Department of Ophthalmology, 2Department of Radiology, 3Department of Medicine, Faculty of Medicine Ramathibodi Hospital, Mahidol University, 4Department of Ophthalmology, Queen Sirikit National Institute of Child Health, Bangkok, Thailand

Abstract: Vertebrobasilar dolichoectasia (VBD) is a rare dilative arteriopathy defined as elongation or widening of the intracranial vertebral and/or basilar arteries. The prevalence ranges from 0.06% to 5.8%. The majority of VBDs are asymptomatic. Downbeat nystagmus has been reported as a component of an infrequent ocular movement disorder in VBD. Nevertheless, oculopalatal tremor (OPT), delayed sequelae of a brainstem lesion, has never been demonstrated in VBD cases. Synchronized rhythmic involuntary contractions of the soft palate with an ocular pendular nystagmus, predominantly vertical pendular nystagmus, are the hallmark presentation. Our case demonstrated a 50-year-old female who presented with 3-month history of oscillopsia. Examination showed binocular conjugate torsional jerk nystagmus simultaneous with a contraction of the soft palate, defined as an atypical OPT, resulting from dolichoectatic left vertebral artery compressing on medulla, demonstrated by magnetic resonance imaging. Bilateral conjugate torsional jerk nystagmus simultaneous with palatal tremor, considering as an atypical OPT, should be included as one of the symptomatic presentations of vertebral artery dolichoectasia in spite of its rarity.

Keywords: vertebral artery dolichoectasia, oculopalatal tremor, torsional nystagmus

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