The ocular tilt reaction (OTR) is a triad of head tilt, cyclotorsion, and skew deviation resulting from unilateral damage to the otolith-ocular pathway [1]. In thalamic infarction, the OTR is known to depend on the involvement of the interstitial nucleus of Cajal (INC), located in the rostral midbrain tegmentum, and not on a lesion in the thalamus itself [2]. Paramedian thalamic infarction involving the INC may cause a contralateral OTR with a subjective visual vertical (SVV) tilt. In contrast, thalamic lesions that do not invade the INC are known to cause SVV tilt without OTR. Here, we present a rare case of isolated complete OTR with unilateral thalamic infarction.
A 71-year-old man presented with a 1-day history of continuous head tilt. He denied having any previous medical illnesses and did not consume alcohol or smoke. Neurological examination revealed skew deviation with hypertrophy of the right eye and head tilt to the left without nystagmus (Fig. 1A). Vertical saccades slowed downward more than upward, and the vertical oculomotor range was limited (up and down approximately 15°); however, the vertical vestibulo-ocular reflex elicited a full range of vertical eye movements. Vertical pursuit was moderately saccadic, more so for the downward pursuit than for the upward. Other neuro-ophthalmological findings were unremarkable. Bedside horizontal head impulse test results were normal. Fundus photography revealed intorsion (6°) of the right eye and extorsion (20°) of the left eye (Fig. 1B). On the SVV tests, the patient had a counterclockwise tilt of 7.66° (normal range, –2.5° to 2.5°; a negative value indicates a counterclockwise tilt). The bithermal caloric tests were symmetrical. Magnetic resonance imaging (MRI), including diffusion-weighted imaging with a 3-mm thin slice, revealed an acute ischemic lesion in the right lateral thalamic infarction, and similar findings were observed for the low-signal lesion on the apparent diffusion coefficient image (Fig. 1C). No additional brainstem or cerebral lesions were observed. Magnetic resonance angiography revealed no steno-occlusion lesions. After 4 days, a follow-up brain MRI showed no additional or extended lesions. One year later, the OTR had completely resolved with a normal SVV tilt.
The main function of the thalamus is to receive sensory information from several sources and distribute it to specific cerebral regions of the brain. The thalamus also contributes to vestibular signal processing and the generation of complex vestibular perceptions. Clinically, an imbalance in vestibular tone at the thalamic level can be demonstrated by special neglect, pusher syndrome, thalamic astasia, and abnormal SVV tilt in isolated thalamic infarction [3,4]. The OTR with SVV tilt occurs in paramedian thalamic infarction because it has been attributed to a common blood supply to the rostral midbrain involving INC [2]. The INC is a prominent group of cells within the medial longitudinal fasciculus of the midbrain, responsible for maintaining vertical eye movement, head posture, and verticality perception [5]. However, no direct lesions were observed in the midbrain tegmentum in our case, despite a complete OTR and an abnormal SVV tilt.
Vestibular signals project to the ventro-intermediate nucleus nuclei (Vim), dorsocaudal nuclei, ventrocaudal posterior and anterior externus, ventrocaudal anterior internus nuclei in the posterolateral part, and the paramedian thalamus. [6]. In addition, electrical stimulation of the Vim in humans evokes rotation or spinning of the body, head, or eyes, either counterclockwise (more commonly) or clockwise [7].
However, Chen and Lin [8] reported an isolated OTR with bilateral paramedian thalamic infarction that did not extend into the rostral midbrain tegmentum. In our case, damage to the posterolateral thalamus, including the Vim, was mainly observed, and the impairment of vertical saccade and contralesional OTR may indicate INC dysfunction. Therefore, we hypothesized that damage to the vestibular subnucleus of the posterolateral or paramedian thalamus, including the Vim, may cause complete OTR, even without extending into the midbrain.
ARTICLE INFORMATION
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Funding/Support
None.
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Conflicts of Interest
Ji-Yun Park is the Editor-in-Chief of Research in Vestibular Science and was not involved in the review process of this article. The authors declare no other conflicts of interest.
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Availability of Data and Materials
All data generated or analyzed during this study are included in this published article. For other data, these may be requested through the corresponding author.
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Authors’ Contributions
Conceptualization, Methodology: JYP; Writing–Original Draft: SJ; Writing–Review & Editing: JYP.
All authors read and approved the final manuscript.
Fig. 1.(A) A left head tilt was observed in this image compared to an old image. (B) Fundus photography reveals binocular ocular torsion in the counterclock wise side (6°intorsion of the right eye and 20°extorsion of left eye). (C) Brain magnetic resonance imaging (MRI) including the axial diffusion-weight image, apparent diffusion coefficient image, and coronal T2-weighted image show acute ischemic stroke in the right thalamus. The scout (yellow line), which indicates the lowest and dorsal part of the lesion on the T1 sagittal MRI, shows no interstitial nucleus of Cajal damage. Written informed consent was obtained for the publication of this letter and accompanying images.
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