Tardive Tourette syndrome in a patient with dementia after 3 weeks' treatment with asenapine.

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The definition of Tourette's disorder in the DSM-5 includes onset before the age of 18 years.1 Adult-onset tics are relatively rare and most cases are secondary.2 Tourette's-disorder-like syndrome after long-term chlorpromazine treatment was first reported in 1978,3 and was named tardive Tourette syndrome (TTS) by Stahl et al. in 1980.4 TTS is characterized by the occurrence of multiple motor and vocal tics in patients on long-term neuroleptic, antiepileptic, or psychostimulant medication.5 Here we report a case of TTS. The patient and his wife provided informed consent for off-label use and publication of this case; the ethics committee of Juntendo University has also approved publication. The case was a 73-year-old male without a known family history of psychiatric disorders. No past or family history of tic disorders was identified. At age 62, he had suffered from depression and was treated for 6 months with antidepressants. Seven years later, he showed tinnitus and hiccup and was referred to a psychiatric hospital. At age 70, he began to show symptoms of dementia; he could not enter numbers on the annual reports for a certified tax accountant and could not cook as he had done previously. Though he was treated with duloxetine and mirtazapine, his symptoms did not improve. He began to show stereotyped, repetitive behaviors, and a lack of sympathy. At age 72, he was treated with clonazepam 0.5 mg/day and quetiapine 25 mg/day. After a month, his repetitive behavior had worsened. He was admitted to a hospital for a detailed examination. The 99mTc-ethyl-cysteinate-dimer single photon emission computed tomography showed significantly decreased blood flow in the temporal and parietal lobes and computed tomography showed mild atrophy of frontal and temporal lobes. Neuropsychological tests showed slight cognitive impairment: the Mini-Mental State Examination, 29/30; the Revised Hasegawa Dementia Scale, 24/30; and the Frontal Assessment Battery, 12/18. He was diagnosed with mild neurocognitive disorder due to Alzheimer's disease according to the DSM-5 criteria1 and treatment with galantamine 8 mg/day was started. After the admission, quetiapine was terminated and off-label use of asenapine 5 mg/day was started despite the lack of solid evidence, which was later increased to 10 mg/day. Within 3 weeks after discharge, he began to manifest a grimace, spit, and repetitive short sound vocalization or shouting. The vocalizations and grimace were affected by situations and he could stop them only for a short period of time and thus we considered them as tics. Therefore, 7 months later, he was admitted to the hospital again and we started to take charge of treatment. Upon admission, he showed preservation, echolalia, and extrapyramidal symptoms. Memantine was started and asenapine and galantamine were stopped. However, his symptoms did not improve. We considered his symptoms as late-onset Tourette's disorder and haloperidol 1 mg/day was added. Three days later, he screamed the whole day, and his tics had worsened. Thus, we reconsidered the symptoms as TTS. Haloperidol was stopped, and quetiapine 100 mg/day and diazepam 4 mg/day were started. After a week, the vocal tics had stopped, but the grimace and mouth movement remained. Four weeks later, quetiapine was stopped, and these symptoms were ameliorated. After the termination of all antipsychotics, he showed disinhibition, preservation, hyperorality, and a lack of sympathy. He was diagnosed as having an early stage of frontotemporal dementia. After discharge, he was treated with memantine 20 mg/day and diazepam 4 mg/day. He joined day care service for senior and enjoyed daily walking. According to a systematic review, TTS was mostly seen in patients with schizophrenia during neuroleptic treatment. Symptoms developed after a few days to 30 years of neuroleptic exposure, or within 1 week to 3 years after discontinuation. Half of the cases had concurrent tardive dyskinesia (TD).5 Recently, Frei et al. proposed the nosology of tardive syndromes resulting from chronic exposure to dopamine receptor blocking agents. The tardive syndromes include TD, tardive dystonia, tardive akathisia, tardive tourettism, tardive myoclonus, tardive chorea, and controversially tardive parkinsonism,6 suggesting that TTS may share its pathophysiology with TD. TTS is thought to be caused by dopamine supersensitivity or blockade of presynaptic dopamine D2 receptor.5 Treatment guidelines for the treatment of tardive syndromes do not conclude the efficacy of withdrawing causative agents or of switching from typical to atypical dopamine receptor blocking agents.7In this case, though galantamine and clonazepam were suggested to be effective for tardive syndromes,7, 8 they were not effective. Diazepam or quetiapine, which is recommended after olanzapine and risperidone,7, 8 was effective. Together with previous reports (shown in Table 1 9, 10), quetiapine might improve dopamine supersensitivity and may be a treatment of choice for TTS. The authors have no conflicts of interest to declare in relation to this report.