| OCR Text |
Show ORIGINAL CONTRIBUTION Ocular Tilt Reaction as a Delayed Complication of Deep Brain Stimulation for Parkinson Disease Santiago Ortiz- Pe ´ rez, MD, Bernardo Sa ´ nchez- Dalmau, MD, Juan Molina, MD, Alfredo Ada ´ n, PhD, Santiago Candela, MD, and Jordi Rumia`, MD Abstract: A 57- year- old- man treated with deep brain stimulation ( DBS) of both subthalamic nuclei for advanced Parkinson disease developed a brain hemorrhage near the site of one of the DBS electrodes 9 months after implantation. The hemor-rhage caused vertical diplopia from skew deviation. Examination also disclosed evidence of ipsiversive binocular torsion and a right head tilt, constituting an ocular tilt reaction ( OTR). Fourteen months later, he was still symptomatic from diplopia. An OTR has not previously been reported as a delayed complication of DBS. ( J Neuro- Ophthalmol 2009; 29: 286- 288) I n patients with advanced Parkinson disease ( PD), deep brain stimulation ( DBS) of the subthalamic nucleus ( STN) has been shown to improve motor function and decrease the need for pharmacologic treatment ( 1- 3). DBS is able to modulate the target region in a reversible and adjustable fashion in contrast to the irreversible and destructive nature of thalamotomy and pallidotomy. With the development of DBS technology and stereotactic neurosurgical techniques, DBS has become a promising therapy not only for PD but also for other disabling movement and neuropsychiatric disorders such as Tourette syndrome, obsessive- compulsive disorder, and refractory depression ( 4). The most frequent side effects documented in relation to DBS are local brain hemorrhage, infection, ischemia, device misplacement, or battery migration ( 5). Aggravated dyskinesias, seizures, and psychiatric disorders have also been reported ( 5,6). Intracerebral hemorrhage generally occurs in the perioperative period but delayed hemorrhage has been described ( 7). A history of hypertension and PD has been reported as a predictive factor ( 8). The ocular tilt reaction ( OTR), a sign of vestibular dysfunction in the roll plane, is characterized by the triad of ocular torsion, skew deviation ( SD), and head tilt ( 9- 11). SD is a vertical strabismus caused by damage to the otolithic- ocular reflex pathway and is associated with abnormal ocular torsion. We describe an OTR as a late complication of DBS. Although conjugate gaze deviation has been reported after STN DBS ( 12,13), we are unaware of previous reports of OTR in this context. CASE REPORT A 54- year- old man with an 8- year stable course of PD developed motor oscillations with on/ off phases uncon-trolled by full doses of levodopa, amantadine, and selegiline. Bilateral DBS of the STN was therefore performed using a surgical procedure reported previously ( 14). In the immediate postoperative period, the patient developed somnolence, disorientation, left hemiparesis, ataxia, and a left Babinski reflex. CT showed an intracerebral hemorrhage adjacent to the right electrode ( Fig. 1A). Three weeks later, the area of hemorrhage had decreased ( Fig. 1B), and all clinical deficits had resolved. DBS provided substantial improvement in the motor signs of PD and in the activities of daily living in this patient, as it has in previously reported patients ( 15). Motor fluctuations and the dose requirements of dopaminergic medication declined, and there were no dyskinesias or other side effects. However, 9 months after implantation of electrodes, the patient suddenly developed vertical diplopia and headache. Our examination disclosed a 20 prism- diopter left hypertropia in primary gaze with a right head tilt ( Fig. 2). Fundus photography showed a left eye incyclo-torsion with perhaps a right eye excyclotorsion ( Fig. 3) ( 16,17). On right gaze, the hypertropia was 8 prism-diopters, and on left gaze it was 30 prism- diopters ( Fig. 2). There was no motor deficit, sensory impairment, ataxia, or change in consciousness. Brain CT revealed a right subthalamic hemorrhage in the same place as the post-operative hemorrhage ( Fig 1C). Institut Clı ´ nic d'Oftalmologı` a ( SO- P, BS- D, JM, AA) and Institut Clı ´ nic de Neurociencies ( SC, JR), Servicio de Neurocirugı ´ a, Hospital Clı ´ nic i Provincial, Barcelona, Spain. Address correspondence to Santiago Ortiz- Perez, c/ Sabino de Arana, ICOF, Hospital Clinic, Sede Maternidad, Barcelona, Spain 08028. E- mail: san. ortiz@ gmail. com 286 J Neuro- Ophthalmol, Vol. 29, No. 4, 2009 The ocular motor findings were interpreted as constituting an ipsiversive OTR, including ocular torsion toward the side of the lesion and SD. No intervention occurred. Fourteen months later, the head tilt and left hypertropia had substantially improved, but the patient still had intermittent diplopia and tilt of the visual environment from the OTR. DISCUSSION We have described a patient who developed a delayed right subthalamic hemorrhage near the site of one of the DBS electrodes. It produced a persistent OTR from which the patient remained symptomatic. The hemorrhage probably resulted from damage to cerebral blood vessels caused by the DBS procedure and the repeated stimulation in the area of the electrode. An OTR is usually attributed to an imbalance in the otolith- ocular and otolith- neck reflexes that are part of a phylogenetically ancient righting response to a lateral tilt of the head. These pathways subserve the vestibulo- ocular reflex ( VOR) ( 18). The primary functions of the VOR are to maintain eye position and stable fixation during head movements ( 9,10,19). Most OTRs are due to a brainstem or cerebellar lesion ( 5,20). The OTR is characterized by perception of tilt of the visual environment, ocular torsion, SD, and a head tilt. The SD is usually comitant but can be incomitant, as in our patient. A binocular conjugate ocular torsion and paradox-ical head tilt ( the head and superior pole of both eyes are rotated toward the hypotropic eye) are typically present ( 9). Unilateral cerebellar or pontomesencephalic lesions affect-ing the interstitial nucleus of Cajal ( INC) or the medial longitudinal fasciculus typically cause contraversive devia-tion ( the contralateral eye is lower). Peripheral vestibular and pontomedullary lesions typically cause ipsiversive deviation ( the ipsilateral eye is lower). Thalamic lesions may cause either contraversive or ipsiversive tilt of the subjective visual vertical ( 9,10,18). The lesion in our patient, which caused an ipsiversive OTR, was located in the right subthalamic region, perhaps affecting the right INC. In humans, mesodiencephalic lesions involving the INC produce a contraversive OTR if the lesion is inhibitory and an ipsiversive OTR if the lesion is excitatory ( 9,20). The bleeding in our patient presumably produced an excitatory injury of the INC. The OTR has been described in association with acute unilateral vestibular lesions ( 21), posterior fossa tumors ( 9), multiple sclerosis ( 9), head trauma ( 9), epileptic FIG. 1. A. Within hours of implantation of a deep brain stimulator, the patient became somnolent and axial CT showed a right subthalamic hematoma ( arrow). B. Three weeks later, the patient had recovered fully, and CT showed that the hematoma had shrunk ( arrow). C. Nine months later, the patient developed an ocular tilt reaction, and CT showed a hematoma in the same region ( arrow). FIG. 2. Ocular alignment at onset of the ocular tilt reaction. In primary gaze position, there is a left hypertropia, which increases on left gaze and reduces on right gaze. The patient also had a right head tilt ( not shown). 287 Ocular Tilt Reaction J Neuro- Ophthalmol, Vol. 29, No. 4, 2009 disorders ( 22), and neurosurgical procedures ( 23). Other isolated documented causes are increased intracranial pressure ( 9), central nervous system malformations ( 9), infections ( 24), cardiac catheterization ( 25), Creutzfeldt- Jakob disease ( 9), polyarteritis nodosa ( 26), and multifocal leukoencephalopathy ( 9). As far as we know, OTR has not previously been reported as a complication of DBS. With the increasing use of such interventions, this possible complication should be recognized, especially because of the participation of the INC in the physiopathology of SD and OTR and its close proximity to the placement of electrodes for DBS treat-ment of PD. REFERENCES 1. The Deep- Brain Stimulation for Parkinson ´ s Disease Study Group. Deep- brain stimulation of the subthalamic nucleus or the pars interna of the globus pallidus in Parkinson's disease. N Engl J Med 2001; 345: 956- 63. 2. Valldeoriola F, Pilleri M, Tolosa E, et al. Bilateral subthalamic stimulation monotherapy in advanced Parkinson ´ s disease: long- term follow- up of patients. Mov Disord 2002; 17: 125- 32. 3. Gan J, Xie- Brustolin J, Mertens P, et al. Bilateral subthalamic nucleus stimulation in advanced Parkinson's disease: three years follow- up. J Neurol 2007; 254: 99- 106. 4. Larson PS. Deep brain stimulation for psychiatric disorders. Neurotherapeutics 2008; 5: 50- 8. 5. Seijo FJ, Alvarez- Vega MA, Gutierrez JC, et al. Complications in subthalamic nucleus stimulation surgery for treatment of Parkinson's disease. Review of 272 procedures. Acta Neurochir ( Wien) 2007; 149: 867- 76. 6. Voges J, Waerzeggers Y, Maarouf M, et al. Deep- brain stimulation: long term analysis of complications caused by hardware and surgery-experiences for a single centre. J Neurol Neurosurg Psychiatry 2006; 77: 868- 72. 7. Zesiewicz TA, Sullivan KL, Hoffmann M, et al. Delayed thalamic intracranial hemorrhage in an essential tremor patient following deep brain stimulation. Eur Neurol 2008; 59: 187- 9. 8. Sansur CA, Frysinger RC, Pouratian N, et al. Incidence of symptomatic hemorrhage after stereotactic electrode placement. J Neurosurg 2007; 107: 998- 1003. 9. Brodsky MC, Donahue SP, Vaphiades M, et al. The skew deviation revisited. Surv Ophthalmol 2006; 51: 105- 28. 10. Dieterich M. Central vestibular disorders. J Neurol 2007; 254: 559- 68. 11. Wong AM, Sharpe JA. Cerebellar skew deviation and the torsional vestibuloocular reflex. Neurology 2005; 65: 412- 9. 12. Krack P, Fraix V, Mendes A, et al. Postoperative management of subthalamic nucleus stimulation for Parkinson's disease. Mov Disord 2002; 17( Suppl 3): 188- 97. 13. Shields DC, Gorgulho A, Behnke E, et al. Contralateral conjugated eye deviation during deep brain stimulation of the subthalamic nucleus. Neurosurgery 2007; 107: 37- 42. 14. Alegret M, Junque ´ C, Valldeoriola F, et al. Effects of bilateral subthalamic stimulation on cognitive function in Parkinson disease. Arch Neurol 2001; 58: 1223- 7. 15. Martı ´ nez- Martı ´ n P, Valldeoriola F, Tolosa E, et al. Bilateral subthalamic nucleus stimulation and quality of life in advanced Parkinson's disease. Mov Disord 2002; 17: 372- 7. 16. Borchert MS. Principles and techniques of the examination ocular motility and alignment. In: Miller NR, Newman NJ, Biousse V, et al., eds. Walsh & Hoyt's Clinical Neuro- Ophthalmology. 6th ed. Philadelphia: Lippincott Williams & Wilkins; 2005; I: 887- 905. 17. Lee H, Yi HA, Lee SR, et al. Ocular torsion associated with infarction in the territory of the anterior inferior cerebellar artery: frequency, pattern, and a major determinant. J Neurol Sci 2008; 269: 18- 23. 18. Zee DS, Newman- Toker D. Supranuclear and internuclear motility disorders. In: Miller NR, Newman NJ, Biousse V, et al., eds. Walsh & Hoyt's Clinical Neuro- Ophthalmology. 6th ed. Philadelphia: Lippincott Williams & Wilkins; 2005; I: 907- 67. 19. Misslisch H, Tweed D. Torsional dynamics and cross- coupling in the human vestibulo- ocular reflex during active head rotation. J Vestib Res 2000; 10: 119- 25. 20. Zackon DS, Sharpe JA. The ocular tilt reaction and skew deviation. In: Sharpe JA, Barber HO, eds. The Vestibulo- ocular Reflex and Vertigo. New York: Raven Press; 1993: 129- 40. 21. Vibert D, Ha ¨ usler R, Safran AB, et al. Diplopia from skew deviation in unilateral peripheral vestibular lesions. Acta Otolaryngol 1996; 116: 170- 6. 22. Galimberti CA, Versino M, Sartori I, et al. Epileptic skew deviation. Neurology 1998; 50: 1469- 72. 23. Riordan- Eva P, Harcourt JP, Faldon M, et al. Skew deviation following vestibular nerve surgery. Ann Neurol 1997; 41: 94- 9. 24. Deibert E, Holland NR. A case of CNS cryptococcosis presenting with slowly alternating skew deviation. Clin Neurol Neurosurg 1996; 98: 192- 3. 25. Suzuki T, Nishio M, Chikuda M, et al. Skew deviation as a complication of cardiac catheterization. Am J Ophthalmol 2001; 132: 282- 3. 26. Ragge NK, Harris CM, Dillon MJ et al. Ocular tilt reaction due to a mesencephalic lesion in juvenile polyarteritis nodosa. Am J Ophthalmol 2003; 135: 249- 51. FIG. 3. Conjugate ocular torsion as evidenced by fundus photography. With the patient in primary gaze position, there is a 15 extorsion of the right eye ( A) and an 8.2 intorsion of the left eye ( B) based on the relationship of the fovea to the optic disc. 288 q 2009 North American Neuro- Ophthalmology Society J Neuro- Ophthalmol, Vol. 29, No. 4, 2009 Ortiz- Pe ´ rez et al |
