Atypical oculopalatal tremor as the presentation of vertebral artery dolichoectasia

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

Isolated Horner syndrome as a rare initial presentation of nasopharyngeal carcinoma: a case report

Tanyatuth Padungkiatsagul,1 Anuchit Poonyathalang,1 Panitha Jindahra,2 Piyaphon Cheecharoen,3 Kavin Vanikieti1 1Department of Ophthalmology, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 2Department of Medicine, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 3Department of Radiology, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand Background: Horner syndrome refers to a set of clinical presentations resulting from disruption of sympathetic innervation to the eye and adnexa. Classically, the clinical triad consists of ipsilateral blepharoptosis, pupillary miosis, and facial anhidrosis. Ocular sympathetic denervation may signify life-threatening causes. Timely investigation and accurate diagnosis are essential in patients with oculosympathetic denervation. Case presentation: A 33-year-old Asian man with a heavy smoking habit presented with a 3-week history of left ptosis and no other complaints. His visual acuity was 20/20 bilaterally. An ophthalmic examination was significant for mild ptosis of his left eyelid and anisocoria (smaller left pupil), which was greater in the dark. Both pupils reacted to light briskly without an afferent pupillary defect. Anhidrosis was found on the medial side of the left forehead. A 10% cocaine test was positive. At his first visit, neurologic examination was unremarkable. Comprehensive radiological investigations were scheduled for a left-sided isolated Horner syndrome. Two weeks after his first visit, he experienced a left-sided headache along with ipsilateral Horner syndrome. Neurologic examination revealed hypoesthesia in the left cranial nerve V1–3 territories. Emergent computed tomography angiography was suspected for petrous part of the left internal carotid artery (ICA) dissection. Magnetic resonance imaging demonstrated an enhancing infiltrative lesion with its epicenter at the left sphenoid bone. The lesion encased the left ICA and invaded the left Meckel cave. Rhinoscopy with incisional biopsy revealed squamous cell nasopharyngeal carcinoma. Conclusion: This case involved an unusual initial presentation of nasopharyngeal carcinoma: isolated Horner syndrome with clinical progression to adjacent structures. Infiltration involving the Meckel cave and ICA at the foramen lacerum can present as postganglionic Horner syndrome associated with trigeminal pain and hypoesthesia. These clinical findings may mimic carotid artery dissection on computed tomography angiography. Detailed magnetic resonance imaging with careful attention to the skull base should be performed. Keywords: Horner syndrome, nasopharyngeal carcinoma, trigemina