Case Report Haloperidol discontinuation in a herpes simplex encephalitis patient with atypical abnormal movements using the herbal medicine Ukgansan-gami: A case report Chul Jin 1 , Jaehak Kim 1 , Woo-Sang Jung, Sang-Kwan Moon, Ki-Ho Cho, Seungwon Kwon* Department of Cardiology and Neurology, College of Korean Medicine, Kyung Hee University, 23 Kyungheedae-ro, Dongdaemun-gu, Seoul 02447, South Korea ARTICLE INFO ABSTRACT Recently, the herbal medicine Ukgansan (Yigansan in China, Yokukansan in Japan) was reported to be effec- tive in the management of movement disorders. We report the case of a 62-year-old woman with herpes simplex encephalitis exhibiting atypical abnormal movements in the chronic stage. While controlling the abnormal movements with haloperidol, an antipsychotic agent, we prescribed Ukgansan-gami, an extract of a variant of Ukgansan, at a dose of 12 g/day to prevent the recurrence of abnormal movements and allow for the discontinuation of haloperidol. The patient was successfully treated with Ukgansan-gami, with no further recurrence of symptoms, making the use of haloperidol no longer necessary. The potential mechanism of action of Ukgansan involves the inhibition of nervous system hyperexcitability through the suppression of glutamate sodium channels, as well as attenuation of hypermotility through serotonin regulation. The pres- ent case suggests that herbal medicine therapy was likely to be an alternative to antipsychotics. © 2019 Elsevier Inc. All rights reserved. Keywords: Ukgansan Yigansan Yokukansan Herpes simplex encephalitis Movement disorder Introduction Herpes simplex encephalitis is caused by the herpes simplex virus type 1, and is the most common type of sporadic encephalitis. 1 It occurs in all age groups, with the highest rate observed in individuals older than 50 years of age. 2 If untreated, the mortality rate is between 60% and 70%. 3 Standard treatment involves the administration of anti- viral agents, usually acyclovir, 3 which reduce the mortality to approxi- mately 28%. 4 However, even with appropriate treatment, the rate of negative outcomes (including death and severe persistent conditions) is as high as 30%. 5 Furthermore, various sequelae can emerge during the recovery phase. Typical consequences are impaired consciousness and symptomatic epilepsy, 3 with amnestic syndrome and abnormal movements often reported. 3 To date, no treatment other than symp- tomatic therapy has been available for these sequelae. Recently, it was reported that the herbal medicine Ukgansan (Yigansan in China, Yokukansan in Japan) may be effective in diseases that involve abnormal movements, such as Huntington disease, 6 rest- less legs syndrome, 7 and tardive dyskinesia. 8 Experimental studies have shown that Ukgansan acts by inhibiting the release of the neu- rotransmitter glutamine. 9À14 We report a case of herpes simplex encephalitis which resulted in atypical abnormal movements in its chronic stage. Abnormal move- ments were successfully controlled in this case with Ukgansan-gami, a variant of Ukgansan, without recurrence. The effectiveness of Ukgansan-gami allowed the treating physicians to discontinue treat- ment with the antipsychotic agent haloperidol, which had been used for controlling the patient’s abnormal movements. Case report A 62-year-old woman was admitted to the neurology department with sudden loss of consciousness, rigidity of limbs, and convulsions. The patient underwent brain magnetic resonance imaging (Fig. 1) and cerebrospinal fluid examination. Based on the findings, she was diagnosed with herpes simplex encephalitis. After the onset, convul- sions persisted and the patient was treated in the intensive care unit. At the end of 74 days of treatment in the intensive care unit, convul- sive seizures improved and the electroencephalographic signatures corresponding to seizures disappeared. However, atypical abnormal movements of the limbs occurred without a determined cause (Fig. 2). The pattern of abnormal movements was as follows: muscle rigidity of the right leg appeared, followed by repeated right leg flex- ion, occurring even if an inspector passively extended the right knee joint. At the same time, the patient was observed involuntarily knocking the bed with alternating legs. * Corresponding author. E-mail addresses: yahaly@naver.com (C. Jin), kimjh6891@gmail.com (J. Kim), skmoon@khu.ac.kr (S.-K. Moon), kkokkottung@hanmail.net (S. Kwon). 1 These authors are equally contributed to this manuscript as first authors https://doi.org/10.1016/j.explore.2019.08.007 1550-8307/© 2019 Elsevier Inc. All rights reserved. Explore 16 (2020) 328À331 Contents lists available at ScienceDirect Explore journal homepage: www.elsevier.com/locate/jsch