The Japanese Journal of Psychiatry and Neurology, Vol. 46, No. 1, 1992
A Case of Tardive Tourette-Like Syndrome Masahiro Kuniyoshi, M.D.," 8 , Kazutoyo Inanaga, M.D.,') g, Katsuyoshi Arikawa, M.D.,S) Yoshiko Maeda, M.D.," * ) Jun Nakamura, M.D." 2, 4, and Naohisa Uchimura, M.D.*) 1 'Chikusuikai Mental Hospital and Clinic, Y a m e "Chikusuikai Institute f o r Neuroinformation, Y a m e 5 ) Kokura Gamou Hospital, Kokura 4 ) Department of Neuropsychiatry, Kirrume University School of Medicine, Kurunre
Abstract: We have had experience in treating tardive Tourette-like syndrome on a chronic schizophrenic patient. The patient was a 38-year-old woman. A diagnosis of schizophrenia was made in 1971 and she received repeated medications for 17 years. In 1989, she began to show vocal tic with coprolalia and motor tic. The medications were haloperidol 18 mg, zotepine 200 mg, levomepromseine 100 mg, biperiden 3 mg and nitrazepam 10 mg at the beginning of Tourette-like syndrome. We have tried to change the medications but this tardive Tourette-like syndrome continued to hang on. However, the symptoms gradually improved after a change in drugs; cessation of biperiden 3 mg and the administration of clonazepam 3 mg. The present case suggested that tardive Tourette-like syndrome might be a subtype of neuroleptic-associated tardive syndromes which might be treated with clonazepam. Key Words:
turdive Tourette-like syndrome, schizophrenia, clonazepam
Jpn J Psychiatr Neurol 46: 67-70, 1992
I NTRODUCT1ON
The importance of antipsychotic drugs in psychiatric treatment has been well established. These drugs provide symptomatic alleviation to psychotic patients, and their social adaptation has been improved. As a result of their widespread use, the side effect, the tardive syndrome, poses problems. Tardive dyskinesia has frequently been reported whereas only a few reports on tardive akathisia, including those dealing with its treatment, have only recently appeared. No __
Received for publication on Sept. 13, 1991. Mailing address: Masahiro Kuniyoshi, M.D., Chikusuikai Mental Hospital and Clinic, Yoshida 1191, Yame City, Fukuoka 834, Japan.
case of manifestation resembling tardive Tourette-like syndrome as in the present case has been reported in Japan. Fewer than 10 such cases have been documented abroad to date. As in the case of tardive dyskinesia, therapy for this condition has yet to be established. A patient with schizophrenia which we had been treating with antipsychotic drugs developed vocal and motor tics resembling those in Gilles de la Tourette syndrome. Various aspects of this condition including etiology are discussed in the following. CASE REPORT The patient was a 38-year-old female
68
M. Kuniyoshi et al.
suffering from schizophrenia. She was the last of three siblings with a normal delivery. She had been brought up with blind Icve. In primary school, she was taciturn and did not attract attention. Her medical history was unremarkable. The family history was negative for psychiatric or neurologic hereditary diseases. In 1971, at 20 years of age, schizophrenia was noted with auditory hallucinations, insomnia, delusions of persecution, and depression. She was hospitalized for about 6 months following the onset, with subsequent admission for about 1.5 months in 1982, from October, 1982, to February, 1984, and from May, 1984, to September, 1985. After the onset, her compliance with instructions fcr drug therapy was excellent. From 1971 to 1984, she was administered haloperidol 8-12 mg, biperiden 3-6 mg, and levomepromazine 50-150 mg with occasional intramuscular haloperidol 5-10 mg as needed for symptomatic aggravation. While being hospitalized from 1984 to 1985, she was given haloperidol 10 mg, biperiden 6 mg, carbamazepine 400 mg, and levomepromazine 50 mg. Following discharge on the same drug dosage regimen, with a slight modification, she regularly attended the outpatient clinic. Cilles de la Tourette-like symptoms complicated her course during the treatment. She attended a day care program held at the local health center and group work without any behavioral problems until around October, 1988, when she began to fail to comply with instructions for drug therapy with complaints of sleepiness and physical dullness. In January, 1989, she stopped attending the day care program and group wcrk. She did not observe meal times. She began to avoid people. She resisted advise, and put an alarm clock in her bed as if the sound of the clock disturbed her. She thought the sound accused her causing her to talk loudly, and not becoming obedient. She became irritab!e and impatient and insomnia had worsened. She was admitted to
th:s hospital on April 24, 1989. On admission, she had auditory hallucinations and was irritative, along with insomnia, impatience, obsessive ideas, delusions of persecution and rejection. Therapy was started with intramuscular and oral haloperidol. This treatment was effective, and she gradually b x a m e calm until around August, 1989, at which time she began to repeat “Sukebe,” an abusive word meaning “lecherous,” and “Chin chin,” another abusive word meaning penis in English. Usage of such language and behavior increased. She told us that she was forced to say what she must not say. At that time, she was on haloperidol 18 mg, zotepine 200 mg, levomepromazine 100 mg, biperiden 3 mg, and nitrazepam 10 mg. In September, her obsessive coprolalia became further pronounced. In addition to the abusive words, she came to repeat “Bakatare,” a word meaning “very foolish,” and “Dobusu,” a word meaning “very ugly.” Her condition resembled a vocal tic in the Gilles de la Tourette syndrome. She began to show a motor tic such as blinking and grimacing. The dose of haloperidol was thus increased to 27 mg, and sultopride 600 mg was given without symptomatic improvement. She began to have asthmatic episodes in the middle of October and thus on November 26, she was transferred to the Department of Psychiatry at Kurume University Hospital for treatment of asthma. Steroid treatment was started to control refractory asthma although transiently. This drug made her restless with aggravation of psychiatric symptoms such as irritation, insomnia, and delusions and so biperiden 3 mg was withdrawn, and clonazepam 3 mg was started in January, 1991 after the alleviation of asthma by medical treatment, Her tics associated with coprolalia subsided gradually, and almost completely disappeared by April. Psychiatric symptoms wzre also alleviated along with improvement of complications. She was transferred to this hospital again. Her subsequent course was uneventful.
A Case of Tardive Tourette-Like Syndrome
69
It is difficult to specify the causative drugs, but haloperidol, zotepine, and levomepromazine may possibly have contributed to The Gilles de la Tourette syndrome is the etiology. Her tics did not improve, and characterized by vocal and motor tics. Acthe psychiatric symptoms became aggravated cording to Moritani", in typical cases, tics even 6 months after the tardive Touretteof the upper half of the body begin at 4-10 like syndrome, despite increased doses of years of age, and gradually spread to the haloperidol along with those of sultopride. lower half. Grinding of the teeth and stickWhen biperiden was discontinued and cloning out the tongue are accompanied by inazcpam 3 mg started, the Tourette-like voluntary barking and meaningless words, as syndrome was alleviated, indicating the same well as echolalia and echopraxia. Eventualmechanism as in tardive dyskinesia to posly, obsessive coprolalia becomes manifest. sibly have been operative at the onset of The pathogenesis has been explained based the present case. Kuniyoshi et a12 stated on organic lesions7 and psychological origin6, clonazepam to be effective for treating but there is no consistent viewpoint. tardive akathisia, and thus the present case The same manifestations as in this Gilles may be one form of the tardive syndrome de la Tourette syndrome were reported in . ~ Tourette-like described by Jeste et ~ 1 This 1978 by Klawans et ~ 1 in : ~psychiatric pais quite obsessive and consequent~ tients on antipsychotic drugs. Jeste et ~ 1 . syndrome ly may frequently be taken as symptomatic reported a total of 7 such cases by 1985. of a psychiatric disease or overlooked due These patients, from 26 to 65 years of age, to concurrent psychological manifestations. consisted of 4 males and 3 females. They Should the present case have been tardive had been on antipsychotic drugs for 3-13 syndrome, the treatment for psychological years before the onset. The major drugs used manifestations would likely have led to an before the onset included chlorpromazine or prolongation of the Touretteaggravation and thioridazine. Their symptoms were noted like syndrome. Careful observation is necesto improve when antipsychotic drugs, halosary for proper treatment. peridol and clonidine were given. This syndrome is considered to arise from excessive REFERENCES activity of the catecholamine system.H Response to drugs resembles that of tardive 1. De Vaugh-Geiss, J.: Tardive Tourette dyskinesia, and thus this tardive Tourettesyndrome. Neurology 30: 526-563, 1980. like syndrome may be a subtype of tardive 2. Jeste, D.V., Wisniewski, A.A. and Wyatt, dyskinesia.2 It is masked by antipsychotic R.J.: Neuroleptic-associated tardive syndromes. Psychiatric Clinics of North Amerdrugs given in large doses and emerges folica 9 133-192, 1986. lowing their withdrawal. However, it re3. Klawans, H.L., Falk, D.K., Nansieda, sponds to therapy better than tardive dysP.A. et al.: Gilles de la Tourette's syndrome kinesia, suggesting that these two conditions after long-term chlorpromazine therapy. may differ in the pathogenetic mechanism.' Neurology 28: 1064-1068, 1978. 4. Kuniyoshi, M., Arikawa, K., Miura, C. In the present case the age of onset suggests and Inanaga, K.: Effect of Clonazepam on that her disease was not the Gilles de la Tardive Akathisia. Human PsychopharmaTourette syndrome. This case may be concology 6: 39-42, 1991. sidered tardive Tourette-like syndrome which 5. Moritani, H.: On Gllles de la Tourette's developed during antipsychotic drug therapy. syndrome. Clinical Psychiatry 19: 61-68, 1977. She had been hospitalized for 17 months 6. Morphew, J.A. and Sim, M.: Gilles de la prior to the onset. Her condition developed Tourette's syndrome: A clinical and psyfollowing treatment vith antipsychotic drugs chopathological study. Brit J Med Psycho1 for 17 years. 42: 293, 1969. DISCUSSION
70
M. Kuniyoshi et al.
7. Shapiro, A.K., Shapiro, E., Wayne, H. and Clarkin, J.: Organic factors in Gilles de la Tourette’s syndrome. Brit J Psychiat 122: 659, 1973.
8. Stahl, S.: Tardive Tourette syndrome in an autistic patient after long-term neuroleptic administration. Am J Psychiat 137: 12671269, 1980.
A Case of Tardive Tourette-Like Syndrome Masahiro Kuniyoshi, M.D.," 8 , Kazutoyo Inanaga, M.D.,') g, Katsuyoshi Arikawa, M.D.,S) Yoshiko Maeda, M.D.," * ) Jun Nakamura, M.D." 2, 4, and Naohisa Uchimura, M.D.*) 1 'Chikusuikai Mental Hospital and Clinic, Y a m e "Chikusuikai Institute f o r Neuroinformation, Y a m e 5 ) Kokura Gamou Hospital, Kokura 4 ) Department of Neuropsychiatry, Kirrume University School of Medicine, Kurunre
Abstract: We have had experience in treating tardive Tourette-like syndrome on a chronic schizophrenic patient. The patient was a 38-year-old woman. A diagnosis of schizophrenia was made in 1971 and she received repeated medications for 17 years. In 1989, she began to show vocal tic with coprolalia and motor tic. The medications were haloperidol 18 mg, zotepine 200 mg, levomepromseine 100 mg, biperiden 3 mg and nitrazepam 10 mg at the beginning of Tourette-like syndrome. We have tried to change the medications but this tardive Tourette-like syndrome continued to hang on. However, the symptoms gradually improved after a change in drugs; cessation of biperiden 3 mg and the administration of clonazepam 3 mg. The present case suggested that tardive Tourette-like syndrome might be a subtype of neuroleptic-associated tardive syndromes which might be treated with clonazepam. Key Words:
turdive Tourette-like syndrome, schizophrenia, clonazepam
Jpn J Psychiatr Neurol 46: 67-70, 1992
I NTRODUCT1ON
The importance of antipsychotic drugs in psychiatric treatment has been well established. These drugs provide symptomatic alleviation to psychotic patients, and their social adaptation has been improved. As a result of their widespread use, the side effect, the tardive syndrome, poses problems. Tardive dyskinesia has frequently been reported whereas only a few reports on tardive akathisia, including those dealing with its treatment, have only recently appeared. No __
Received for publication on Sept. 13, 1991. Mailing address: Masahiro Kuniyoshi, M.D., Chikusuikai Mental Hospital and Clinic, Yoshida 1191, Yame City, Fukuoka 834, Japan.
case of manifestation resembling tardive Tourette-like syndrome as in the present case has been reported in Japan. Fewer than 10 such cases have been documented abroad to date. As in the case of tardive dyskinesia, therapy for this condition has yet to be established. A patient with schizophrenia which we had been treating with antipsychotic drugs developed vocal and motor tics resembling those in Gilles de la Tourette syndrome. Various aspects of this condition including etiology are discussed in the following. CASE REPORT The patient was a 38-year-old female
68
M. Kuniyoshi et al.
suffering from schizophrenia. She was the last of three siblings with a normal delivery. She had been brought up with blind Icve. In primary school, she was taciturn and did not attract attention. Her medical history was unremarkable. The family history was negative for psychiatric or neurologic hereditary diseases. In 1971, at 20 years of age, schizophrenia was noted with auditory hallucinations, insomnia, delusions of persecution, and depression. She was hospitalized for about 6 months following the onset, with subsequent admission for about 1.5 months in 1982, from October, 1982, to February, 1984, and from May, 1984, to September, 1985. After the onset, her compliance with instructions fcr drug therapy was excellent. From 1971 to 1984, she was administered haloperidol 8-12 mg, biperiden 3-6 mg, and levomepromazine 50-150 mg with occasional intramuscular haloperidol 5-10 mg as needed for symptomatic aggravation. While being hospitalized from 1984 to 1985, she was given haloperidol 10 mg, biperiden 6 mg, carbamazepine 400 mg, and levomepromazine 50 mg. Following discharge on the same drug dosage regimen, with a slight modification, she regularly attended the outpatient clinic. Cilles de la Tourette-like symptoms complicated her course during the treatment. She attended a day care program held at the local health center and group work without any behavioral problems until around October, 1988, when she began to fail to comply with instructions for drug therapy with complaints of sleepiness and physical dullness. In January, 1989, she stopped attending the day care program and group wcrk. She did not observe meal times. She began to avoid people. She resisted advise, and put an alarm clock in her bed as if the sound of the clock disturbed her. She thought the sound accused her causing her to talk loudly, and not becoming obedient. She became irritab!e and impatient and insomnia had worsened. She was admitted to
th:s hospital on April 24, 1989. On admission, she had auditory hallucinations and was irritative, along with insomnia, impatience, obsessive ideas, delusions of persecution and rejection. Therapy was started with intramuscular and oral haloperidol. This treatment was effective, and she gradually b x a m e calm until around August, 1989, at which time she began to repeat “Sukebe,” an abusive word meaning “lecherous,” and “Chin chin,” another abusive word meaning penis in English. Usage of such language and behavior increased. She told us that she was forced to say what she must not say. At that time, she was on haloperidol 18 mg, zotepine 200 mg, levomepromazine 100 mg, biperiden 3 mg, and nitrazepam 10 mg. In September, her obsessive coprolalia became further pronounced. In addition to the abusive words, she came to repeat “Bakatare,” a word meaning “very foolish,” and “Dobusu,” a word meaning “very ugly.” Her condition resembled a vocal tic in the Gilles de la Tourette syndrome. She began to show a motor tic such as blinking and grimacing. The dose of haloperidol was thus increased to 27 mg, and sultopride 600 mg was given without symptomatic improvement. She began to have asthmatic episodes in the middle of October and thus on November 26, she was transferred to the Department of Psychiatry at Kurume University Hospital for treatment of asthma. Steroid treatment was started to control refractory asthma although transiently. This drug made her restless with aggravation of psychiatric symptoms such as irritation, insomnia, and delusions and so biperiden 3 mg was withdrawn, and clonazepam 3 mg was started in January, 1991 after the alleviation of asthma by medical treatment, Her tics associated with coprolalia subsided gradually, and almost completely disappeared by April. Psychiatric symptoms wzre also alleviated along with improvement of complications. She was transferred to this hospital again. Her subsequent course was uneventful.
A Case of Tardive Tourette-Like Syndrome
69
It is difficult to specify the causative drugs, but haloperidol, zotepine, and levomepromazine may possibly have contributed to The Gilles de la Tourette syndrome is the etiology. Her tics did not improve, and characterized by vocal and motor tics. Acthe psychiatric symptoms became aggravated cording to Moritani", in typical cases, tics even 6 months after the tardive Touretteof the upper half of the body begin at 4-10 like syndrome, despite increased doses of years of age, and gradually spread to the haloperidol along with those of sultopride. lower half. Grinding of the teeth and stickWhen biperiden was discontinued and cloning out the tongue are accompanied by inazcpam 3 mg started, the Tourette-like voluntary barking and meaningless words, as syndrome was alleviated, indicating the same well as echolalia and echopraxia. Eventualmechanism as in tardive dyskinesia to posly, obsessive coprolalia becomes manifest. sibly have been operative at the onset of The pathogenesis has been explained based the present case. Kuniyoshi et a12 stated on organic lesions7 and psychological origin6, clonazepam to be effective for treating but there is no consistent viewpoint. tardive akathisia, and thus the present case The same manifestations as in this Gilles may be one form of the tardive syndrome de la Tourette syndrome were reported in . ~ Tourette-like described by Jeste et ~ 1 This 1978 by Klawans et ~ 1 in : ~psychiatric pais quite obsessive and consequent~ tients on antipsychotic drugs. Jeste et ~ 1 . syndrome ly may frequently be taken as symptomatic reported a total of 7 such cases by 1985. of a psychiatric disease or overlooked due These patients, from 26 to 65 years of age, to concurrent psychological manifestations. consisted of 4 males and 3 females. They Should the present case have been tardive had been on antipsychotic drugs for 3-13 syndrome, the treatment for psychological years before the onset. The major drugs used manifestations would likely have led to an before the onset included chlorpromazine or prolongation of the Touretteaggravation and thioridazine. Their symptoms were noted like syndrome. Careful observation is necesto improve when antipsychotic drugs, halosary for proper treatment. peridol and clonidine were given. This syndrome is considered to arise from excessive REFERENCES activity of the catecholamine system.H Response to drugs resembles that of tardive 1. De Vaugh-Geiss, J.: Tardive Tourette dyskinesia, and thus this tardive Tourettesyndrome. Neurology 30: 526-563, 1980. like syndrome may be a subtype of tardive 2. Jeste, D.V., Wisniewski, A.A. and Wyatt, dyskinesia.2 It is masked by antipsychotic R.J.: Neuroleptic-associated tardive syndromes. Psychiatric Clinics of North Amerdrugs given in large doses and emerges folica 9 133-192, 1986. lowing their withdrawal. However, it re3. Klawans, H.L., Falk, D.K., Nansieda, sponds to therapy better than tardive dysP.A. et al.: Gilles de la Tourette's syndrome kinesia, suggesting that these two conditions after long-term chlorpromazine therapy. may differ in the pathogenetic mechanism.' Neurology 28: 1064-1068, 1978. 4. Kuniyoshi, M., Arikawa, K., Miura, C. In the present case the age of onset suggests and Inanaga, K.: Effect of Clonazepam on that her disease was not the Gilles de la Tardive Akathisia. Human PsychopharmaTourette syndrome. This case may be concology 6: 39-42, 1991. sidered tardive Tourette-like syndrome which 5. Moritani, H.: On Gllles de la Tourette's developed during antipsychotic drug therapy. syndrome. Clinical Psychiatry 19: 61-68, 1977. She had been hospitalized for 17 months 6. Morphew, J.A. and Sim, M.: Gilles de la prior to the onset. Her condition developed Tourette's syndrome: A clinical and psyfollowing treatment vith antipsychotic drugs chopathological study. Brit J Med Psycho1 for 17 years. 42: 293, 1969. DISCUSSION
70
M. Kuniyoshi et al.
7. Shapiro, A.K., Shapiro, E., Wayne, H. and Clarkin, J.: Organic factors in Gilles de la Tourette’s syndrome. Brit J Psychiat 122: 659, 1973.
8. Stahl, S.: Tardive Tourette syndrome in an autistic patient after long-term neuroleptic administration. Am J Psychiat 137: 12671269, 1980.
