Accepted Manuscript Transient blepharospasm, apraxia of eyelid opening, and hemidyskinesia following a right parietotemporal infarct Cheng-Yang Hsieh , Pi-Shan Sung , Wen-Juh Hwang , MD PII: S1353-8020(14)00215-6 DOI: 10.1016/j.parkreldis.2014.05.012 Reference: PRD 2351 To appear in: Parkinsonism and Related Disorders Received Date: 8 February 2014 Revised Date: 10 May 2014 Accepted Date: 25 May 2014 Please cite this article as: Hsieh C-Y, Sung P-S, Hwang W-J, Transient blepharospasm, apraxia of eyelid opening, and hemidyskinesia following a right parietotemporal infarct, Parkinsonism and Related Disorders (2014), doi: 10.1016/j.parkreldis.2014.05.012. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. 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ACCEPTED MANUSCRIPT “Video is part of the manuscript” Transient blepharospasm, apraxia of eyelid opening, and hemidyskinesia Cheng-Yang Hsieha,b, Pi-Shan Sungc, Wen-Juh Hwangd RI PT following a right parietotemporal infarct Department of Neurology, Sin Lau Hospital, Tainan, Taiwan b Institute of Biopharmaceutical Sciences, College of Medicine, National Cheng Kung University, Tainan, Taiwan c M AN U SC a Department of Neurology, National Cheng Kung University Hospital, Tainan, Taiwan d Department of Neurology, National Cheng Kung University Hospital, College of TE D Medicine, National Cheng Kung University, Tainan, Taiwan EP Corresponding author: AC C Wen-Juh Hwang, MD Department of Neurology National Cheng Kung University Hospital College of Medicine, National Cheng Kung University 138 Sheng-Li Road, Tainan 704, Taiwan Tel: +886-6-276-6187; Fax: +886-6-237-4285; e-mail: wjhwang@mail.ncku.edu.tw 1 ACCEPTED MANUSCRIPT Word counts: Text: 760; References: 5; Tables: 0; Figures: 1; Video: 1 Funding, sponsorship: No corporate, government, or institutional funding. RI PT Relevant conflicts of interest/financial disclosures: Nothing to report. Authors’ roles: SC Cheng-Yang Hsieh: Wrote first draft; AC C EP TE D M AN U Pi-Shan Sung and Wen-Juh Hwang: Reviewed and critiqued the manuscript. 2 ACCEPTED MANUSCRIPT Blepharospasm and apraxia of eyelid opening have rarely been reported in acute nondominant hemispheric stroke [1,2]. It is also rare for hyperkinetic movement RI PT disorders to develop after acute hemispheric stroke [3]. We report a patient with an acute right parietotemporal infarct who presented with transient blepharospasm, apraxia of eyelid opening, and left hemidyskinesia. SC A 71-year-old right-handed man was admitted with acute onset of involuntary M AN U movement in his left limbs and face, and difficulty opening his eyes. He had a medical history of hypertension, atrial fibrillation, rheumatic heart disease after mechanical mitral valve replacement, and congestive heart failure. He was treated with digoxin, furosemide, and warfarin. Neurological examinations showed normal TE D orientation and normal speech, without aphasia or dysarthria. He had right-to-left disorientation, left hemineglect, left homonymous hemianopia, visuospatial EP impairment, and mild left hemiparesis, but no asomatognosia, astereognosia, or AC C anosognosia. His pupils, ocular motility, somatosensory system (evaluated by pinprick and sense of passive movement), cerebellar system, and sphincters were normal. The patient showed intermittent forceful contraction of the bilateral orbicularis oculi and corrugator muscles. He was also unable to open his eyes at will despite vigorous forceful contraction of the frontalis muscles and in the absence of orbicularis oculi contraction. There were frequent intermittent, irregular, fast, and more or less 3 ACCEPTED MANUSCRIPT patterned movements in his left limbs and face in the wakeful state (Video). His left hand was continually grasping. He did not display compulsive exploration of the RI PT environment or intermanual conflict. The patient did not show any avoidance posture or levitation in his left upper limb. A brain computed tomography (not shown) and magnetic resonance imaging (Fig. SC 1) showed acute infarction in the right parietotemporal lobe. The basal ganglia, M AN U brainstem, and thalamus were normal. The dyskinetic movement responded well to intravenous diphenhydramine (30 mg): it completely disappeared in 10 minutes and recurred 4 hours later. He was then treated with haloperidol (15 mg/day). Both eyelid abnormalities and left hemidyskinesia disappeared in 4 days. The dose of haloperidol TE D was gradually tapered and discontinued 10 days after its initiation, without a recurrence of the eyelid abnormalities or left hemidyskinesia. EP The temporal lobe regulates memory, emotions, impulse control (sex, eating), AC C hearing, smell, language, and learning. The amygdala in the temporal lobe plays a major role in processing unconscious emotional facial expression. Currently, there is no neurobiological evidence to support a link between the temporal lobe and the motor control of eyelids and limbs through the pyramidal tract [4]. Because the temporal lobe does not contribute to the pyramidal tract, the amygdala was not involved in our patient, and because the patient was conscious of his involuntary 4 ACCEPTED MANUSCRIPT movement, we proposed that the eyelid abnormality and hemidyskinesia were related to the parietal lobe lesion. RI PT Parietal stroke rarely presents with hemiballism-hemichorea (HB-HC) [5], alien hand phenomenon, or action or delayed dystonia. The left hemidyskinesia in our patient is different from previous observations of patients with hemidystonia, which SC usually develops several months to years after an acute insult. Hemidystonia is M AN U usually refractory to treatment and persists after its appearance. The more or less patterned movement and a predominant involvement of the face and upper limb are different from HB-HC. “Pseudochoreoathetosis” is also unlikely, because our patient had no sensory abnormalities. The involvement of the face, the continual left hand TE D grasping behavior, and the lack of avoidance response or levitation, did not favor posterior alien hand syndrome. We speculate that the parietal lobe lesion caused EP ignition failure for the voluntary eyelid opening (apraxia of eyelid opening) and AC C disinhibition of basal ganglia and brainstem interneurons with the manifestations of blepharospasm and left hemidyskinesia. Diphenhydramine is an anticholinergic antihistamine effective for treating acute akathisia and acute dystonic reaction. The sedative effect of antihistamine may suppress movement disorders. However, our patient remained clear and his hemidyskinesia disappeared after diphenhydramine treatment. N-methyl-D-aspartate 5 ACCEPTED MANUSCRIPT (NMDA) receptor antagonists such as amantadine alleviate dyskinesia in patients with Parkinson’s disease. Because diphenhydramine, a histamine-1 receptor antagonist, has RI PT not been reported to affect NMDA receptors, the clinical effect is not likely due to an interaction with the glutaminergic pathway. Determining how diphenhydramine suppresses the dyskinesia in this patient is further complicated by the fact that the SC mechanism of hemidyskinesia in not fully understood. The paradoxical response of M AN U hyperkinetic movement disorders both to anti-cholinergic and to anti-dopaminergic agents was unclear. This report has several limitations. First, we did not have simultaneous electromyographic recordings from the levator palpebrae superioris and orbicularis TE D oculi muscles. Second, we did not have a blink reflex to suggest hyperexcitability of the brainstem interneurons. Third, we lacked perfusion imaging in the acute stage to AC C EP exclude basal ganglia hypoperfusion. 6 ACCEPTED MANUSCRIPT References [1] Averbuch-Heller L, Leigh RJ, Mermelstein V, Zagalsky L, Streifler JY. Ptosis in RI PT patients with hemispheric strokes. Neurology 2002;58:620–4. [2] Jacob PC, Chand RP. Blepharospasm and jaw closing dystonia after parietal infarcts. Mov Disord 1995;19:794–5. SC [3] Ghika-Schmid F, Ghika J, Regli F, Bogousslavsky J. Hyperkinetic movement M AN U disorders during and after acute stroke: the Lausanne Stroke Registry. J Neurol Sci 1997;146:109–16. [4] Toyoshima K, Sakai H. Exact cortical extent of the origin of the corticospinal tract (CST) and the quantitative contribution to the CST in different cytoarchitectonic areas: TE D A study with horseradish peroxidase in the monkey. J Hirnforsch 1982;23:257–69. [5] Mizushima N, Park-Matsumoto YC, Amakawa T, Hayashi H. A case of AC C 1997;37:65–6. EP hemichorea-hemiballism associated with parietal lobe infarction. Eur Neurol 7 ACCEPTED MANUSCRIPT Legend to the Video The video shows blepharospasm, apraxia of eyelid opening, and hemidyskinesia in RI PT the left limbs and face. The patient is asked to open and close his hands in succession: right hand first and then the left hand. He shows right-to-left disorientation when he is asked to hold up one finger of the left hand. When he is asked to use his left hand to SC touch his right ear, his left hand went to his left ear first without touching it, and then M AN U moved to touch his right ear. The left hemidyskinesia is not aggravated during the AC C EP TE D voluntay movements. 8 ACCEPTED MANUSCRIPT FIGURE LEGEND Fig. 1. RI PT A diffusion-weighted magnetic resonance image (MRI) (left column) showing restricted diffusion in the right parietotemporal lobe. A T2-weighted MRI (right column) showing high signal intensity in the corresponding area. The midbrain, SC subthalamic nucleus, striatum, globus pallidus, and thalamus were normal in this AC C EP TE D M AN U study. The image shows acute infarction in the right parietotemporal lobe. 9 AC C EP TE D M AN U SC RI PT ACCEPTED MANUSCRIPT