Neurocase The Neural Basis of Cognition ISSN: 1355-4794 (Print) 1465-3656 (Online) Journal homepage: http://www.tandfonline.com/loi/nncs20 Akinetic mutism reversibility after L-dopa therapy in unilateral left anterior cerebral artery infarction Guery Deborah, Elodie Ong & Norbert Nighoghossian To cite this article: Guery Deborah, Elodie Ong & Norbert Nighoghossian (2017): Akinetic mutism reversibility after L-dopa therapy in unilateral left anterior cerebral artery infarction, Neurocase, DOI: 10.1080/13554794.2017.1319493 To link to this article: http://dx.doi.org/10.1080/13554794.2017.1319493 Published online: 27 Apr 2017. Submit your article to this journal Article views: 2 View related articles View Crossmark data Full Terms & Conditions of access and use can be found at http://www.tandfonline.com/action/journalInformation?journalCode=nncs20 Download by: [The UC San Diego Library] Date: 01 May 2017, At: 18:31 NEUROCASE, 2017 https://doi.org/10.1080/13554794.2017.1319493 LETTER Akinetic mutism reversibility after L-dopa therapy in unilateral left anterior cerebral artery infarction Guery Deborah, Elodie Ong and Norbert Nighoghossian Stroke Unit, Hôpital Neurologique Pierre Wertheimer, Hospices Civils de Lyon, Université Claude Bernard Lyon 1, Lyon, France Dear editor Akinetic mutism is a neurological disorder characterized by diminished voluntary movement, speech, and thought, without disturbance of arousal. This syndrome has been rarely reported following unilateral anterior cerebral artery infarction. Functional interruption of the dopaminergic pathways may support clinical symptoms. Clinical reversibility after the introduction of L-dopa therapy is poorly documented. A 61-year-old woman with chronic high blood pressure presented with an acute right-sided weakness. Neurological examination revealed a right hemiplegia, a severe mutism, and a global akinesia. She had a grasp reflex with forced grasping. Magnetic resonance imaging revealed a left anterior cerebral artery stroke involving the sensory, motor, supplementary motor area, prefrontal, and anterior part of the cingulate gyrus. Magnetic resonance angiography revealed a right anterior cerebral artery occlusion with an A1 segment hypoplasia (Figure 1a(,b)). L-dopa was introduced 4 days later (62.5 mg three times/daily) and she recovered gradually, and at day 7, her speech was still impaired but she was able to repeat small sentences, to give her name and address. She was less catatonic but motor and sensory symptoms persisted. Two weeks later, she was more fluent despite episodic echolalia and semantic paraphasias. Akinetic mutism is classically related to bilateral infarction within anterior cerebral artery territory, and unilateral involvement is uncommon. Akinesia and mutism with restriction of spontaneous activity depend on cingulate gyrus or supplementary motor area lesion (Alexander, Delaney, & Strick, 1990; Combarros, Infante, & Berciano, 2000; Nagaratnam, Nagaratnam, Ng, & Diu, 2004). This case suggests that the akinetic mutism caused by anterior cerebral arterial infarct is closely linked to the perturbation of the subcortical dopaminergic system and the amelioration of the clinical features concordantly evolved with the restoration of the dopaminergic function. Neurological improvement after the introduction of L-dopa in akinetic mutism due to anterior cerebral artery infarction is poorly documented. This improvement may be inconstant but affects the various symptoms related to the different circuits involved in neurobehavioral changes (Floel, Hummel, Breitenstein, Knecht, & Cohen, 2005). Moreover, studies showed that levodopa in patients with chronic stroke enhanced motor memory formation (Seniów, Litwin, Litwin, Leśniak, & Czonkowska, 2009) and may improve language impairment, particularly in patients with anterior lesions (Yang et al., 2007). Although therapeutic data are limited in the absence of controlled studies, so far we have identified three cases of unilateral anterior cerebral artery infarction. A favorable clinical course was observed in all cases to a varying degree. The response was obtained at different dosages of levodopa. This drug seems well tolerated and likely underused in the presence of this clinical syndrome where few medical alternatives are available. Disclosure statement The authors declare that they have no conflict of interest. References Figure 1. Magnetic resonance imaging flair sequence: Left anterior cerebral artery stroke involving the sensory, motor, supplementary motor area and cingular cortex. CONTACT Norbert Nighoghossian norbert.nighoghossian@chu-lyon.fr © 2017 Informa UK Limited, trading as Taylor & Francis Group Alexander, G. E., Delaney, M. R., & Strick, P. L. (1990). Parallel organisations of functionally segregated circuits linking basal ganglia and cortex. Annals Reviews Neuroscience, 9, 357–381. doi:10.1146/annurev. ne.09.030186.002041 Combarros, O., Infante, J., & Berciano, J. (2000). Akinetic mutism from frontal lobe damage responding to levodopa. Journal of Neurology, 247, 568–569. doi:10.1007/s004150070161 2 G. DEBORAH ET AL. Floel, A., Hummel, F., Breitenstein, C., Knecht, S., & Cohen, L. G. (2005). Dopaminergic effects on encoding of a motor memory in chronic stroke. Neurology, 65, 472–474. doi:10.1212/01.wnl.0000172340.56307.5e Nagaratnam, N., Nagaratnam, K., Ng, K., & Diu, P. J. (2004). Akinetic mutism following stroke. Journal of Clinical Neuroscience, 11, 25–30. doi:10.1016/j.jocn.2003.04.002 Seniów, J., Litwin, M., Litwin, T., Leśniak, M., & Czonkowska, A. (2009). A.new approach to the rehabilitation of post-stroke focal cognitive syndrome: Effect of levodopa combined with speech and language therapy on functional recovery from aphasia. Journal of the Neurological Sciences, 283, 214– 218. doi:10.1016/j.jns.2009.02.336 Yang, C. P., Huang, W. S., Shih, H. T., Lin, C. Y., Lu, M. K., Kao, C. H., . . . Tsai, C. H. (2007). Diminution of basal ganglia dopaminergic function may play an important role in the generation of akinetic mutism in a patient with anterior cerebral arterial infarct. Clinical Neurology and Neurosurgery, 109, 602–606. doi:10.1016/j.clineuro.2007.04.012