- Email: [email protected]
Chronic Pisa syndrome associated with switching antipsychotics from olanzapine to ziprasidone
Progress in Neuro-Psychopharmacology & Biological Psychiatry 33 (2009) 162–163
Contents lists available at ScienceDirect
Progress in Neuro-Psychopharmacology & Biological Psychiatry j o u r n a l h o m e p a g e : w w w. e l s e v i e r. c o m / l o c a t e / p n p b p
Letter to the Editor (Case report)
Chronic Pisa syndrome associated with switching antipsychotics from olanzapine to ziprasidone Yi-Wei Yeh a, Yi-Chyan Chen a, Chih-Kang Chen a, Hui-Ming Feng b, Tzu-Yun Wang a, Chih-Lun Chen a,⁎ a b
Department of Psychiatry, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan Division of Medicine, National Defense Medical Center, Taipei, Taiwan
1. Introduction Pisa syndrome (PS), so called pleurothotonus, a rare asymmetric axial dystonia characterized by tonic lateral flexion of the trunk, has been mainly observed in patients using typical antipsychotics (Stubner et al., 2000; Suzuki and Matsuzaka, 2002). It also developed in rare cases using atypical antipsychotics and in patients with neurodegenerative disorder such as Alzheimer's disease or Parkinson's disease (Yassa et al., 1991; Suzuki and Matsuzaka, 2002). Ignoring PS will decrease patients' ability to participate in therapy and rehabilitation and will influence their quality of life. Here we present a patient with schizophrenia, who developed chronic PS for 2 years after switching antipsychotics from olanzapine to ziprasidone; the dystonia vanished after the cessation of ziprasidone.
2. Case report A 38-year-old single unemployed male had a history of paranoid type schizophrenia (meeting DSM-IV criteria) for 15 years. He had no history of perinatal or developmental abnormality, general medical disease or drug abuse. From 1993 to 2005, three different antipsychotics were used successively for 4–5 years each, including methylperidol 20 mg/day, sulpiride 600 mg/day, and olanzapine 10–20 mg/day. Weight gain of 25 kg, increase in waist circumference by 18 cm, and dyslipidemia were found during 2002–2005 under olanzapine treatment. To avoiding worsening of the metabolic syndrome, the antipsychotic was switched to ziprasidone 160 mg/day in July 2005 in acute ward and maintained in day-care ward during 2005–2007. However, one month after using ziprasidone, the patient began leaning to one side. This change progressed to the point where he was tilted at an angle of 25–30°. The leaning posture was persistent and refractory to anticholinergic (trihexyphenidyl 5 mg/day) administrated under the initial impression of tardive dystonia. The patient seemed unaware of and unconcerned about this change. He did not receive any change on his medication in day hospitalization until August 2007 when he was admitted to our hospital because of a psychotic episode. A posture of tonic flexion of his trunk and tilting his
⁎ Corresponding author. Department of Psychiatry, Tri-Service General Hospital, No. 325, Sec. 2, Cheng-Kung Road, Nei-Hu Area, Taipei 114, Taiwan. Tel.: +11 886 2 26739211; fax: +11 886 2 87927221. E-mail address: [email protected] (C.-L. Chen). 0278-5846/$ – see front matter © 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.pnpbp.2008.11.001
head to the right side while sitting, standing, and walking was observed. He showed no other signs or symptoms suggestive of other EPS. The differential diagnoses of hysterical and catatonic presentation were both ruled out. Brain CT and EMG did not reveal any abnormality. Pisa syndrome was diagnosed. After switching to amisulpride 600 mg/ day for one week, the dystonia resolved completely without adjuvant treatment. Two weeks later, his psychotic symptoms also improved (CGI-S: 4–5, CGI-I: 2–3). At 12 months follow-up, the patient did not manifest any motor complication. 3. Discussion Ziprasidone, a serotonin-dopamine antagonist, has higher 5-HT2A/ D2 ration among atypical antipsychotics, and it is suggested that it has a lower risk of EPS (Schmidt et al., 2001; Dayalu and Chou, 2008). However, Pisa syndrome (PS) is a rare involuntary movement disorder and is distinguishable from other EPS. First, PS occurs mostly during the first month of treatment with atypical antipsychotics, whereas acute dystonia occurs within the first 24 h of treatment (Dayalu and Chou, 2008). Only 40% of reported PS responded to treatment by anticholinergics (Suzuki and Matsuzaka, 2002); its response is better than tardive dystonia but inferior to acute dystonia. PS mostly vanished after the discontinuation or reduction of the offending drug without any adjunctive treatment (Stubner et al., 2000), but tardive dystonia might persist (Dayalu and Chou, 2008). Consistent with previous reports, PS developed one month after using ziprasidone and persisted for 2 years under maintenance treatment; it resolved entirely upon discontinuation of ziprasidone even after a two-year course. Such a cause-and-effect relationship between the use of ziprasidone and the appearance of PS implied ziprasidone-induced PS (the Naranjo causality-scale score was 6). Although most evidence supports the possible pathophysiology of dopaminergic–cholinergic imbalance in development of Pisa syndrome (Stubner et al., 2000; Suzuki and Matsuzaka, 2002; Villarejo et al., 2003; Duggal et al., 2004), a serotonin–norepinephrine effect as well as dopamine interaction as causes of PS development should nevertheless be taken into account (Remington, 1988; Suzuki et al., 1997; Suzuki and Matsuzaka, 2002), particularly for ziprasidone with its rich pharmacologic profile of moderate serotonin–norepinephrine reuptake inhibitor, 5-HT1A agonism, and 5-HT1B/D, 5-HT2A/2C antagonism (Schmidt et al., 2001). However, the possible causality of PS might be specific to the introduction of a new drug (Suzuki and Matsuzaka, 2002), the
Letter to the Editor (Case report)
discontinuation of a former drug (Stubner et al., 2000), or the process of drug switching (Duggal et al., 2004; Rota et al., 2007). To date, only two cases of ziprasidone-associated PS have been reported. Ziegenbein et al. (2003) reported that a 38-year-old schizophrenic woman developed PS on day 18 while switching from clozapine 275 mg/day to ziprasidone 80 mg/day. The PS disappeared after withdrawal of ziprasidone and switching to amisulpride 14 days later. Duggal (2008) described a 20-year-old man with schizoaffective disorder comorbid with cocaine dependence. The patient showed acute pharyngolaryngeal dystonia and PS after administration of ziprasidone 120 mg/ day and valproate 1000 mg/day on day 2. The dystonia resolved after addition of anticholinergics. The author did not indicate the psychotropic medication prior to ziprasidone and could not exclude the possible effect of valproate (Yohanan et al., 2006; Duggal, 2008). Coincidentally, our case is similar to that reported by Ziegenbein et al. (2003), in that PS developed during atypical antipsychotic treatment after switching from one drug to another. In contrast to ziprasidone with its weak anticholinergic properties, both clozapine and olanzapine have potent anticholinergic effects. Switching antipsychotics with different anticholingeric effects could increase the risk of a cholinergic rebound (Lambert, 2007) and might result in dopaminergic-cholinergic imbalance. From this point of view, switching antipsychotics maybe a potential risk factor for development of PS (Duggal et al., 2004; Rota et al., 2007) should be performed slowly and gradually, especially when the former antipsychotic has a potent anticholinergic effect. 4. Conclusion Clinicians and patients easily neglected the PS. At least one study had shown that only 25% patients with schizophrenia are aware of their involuntary movements (Caracci et al., 1990) and that this was
163
correlated with their insight level. Clinicians have to keep in mind this motor complication to avoid worsening of symptoms and resulting in iatrogenic accidents. References Caracci G, Mukherjee S, Roth SD, Decina P. Subjective awareness of abnormal involuntary movements in chronic schizophrenic patients. Am J Psychiatry 1990;147:295–8. Dayalu P, Chou KL. Antipsychotic-induced extrapyramidal symptoms and their management. Expert Opin Pharmacother 2008;9:1451–62. Duggal HS. Acute Pisa syndrome and pharnygolaryngeal dystonia due to ziprasidone. J Neuropsychiatry Clin Neurosci 2008;20:108–9. Duggal HS, Sivamony S, Umapathy C. Pisa syndrome and atypical antipsychotics. Am J Psychiatry 2004;161:373. Lambert TJ. Switching antipsychotic therapy: what to expect and clinical strategies for improving therapeutic outcomes. J Clin Psychiatry 2007;68(Suppl 6):10–3. Remington GJ. The Pisa syndrome: possible role for serotonin and noradrenaline. J Clin Psychopharmacol 1988;8:228–9. Rota E, Bergesio G, Dettoni E, Demicheli CM. Pisa syndrome during aripiprazole treatment: a case report. Prog Neuropsychopharmacol Biol Psychiatry 2007;31:286–7. Schmidt AW, Lebel LA, Howard Jr HR, Zorn SH. Ziprasidone: a novel antipsychotic agent with a unique human receptor binding profile. Eur J Pharmacol 2001;425:197–201. Stubner S, Padberg F, Grohmann R, Hampel H, Hollweg M, Hippius H, et al. Pisa syndrome (pleurothotonus): report of a multicenter drug safety surveillance project. J Clin Psychiatry 2000;61:569–74. Suzuki T, Matsuzaka H. Drug-induced Pisa syndrome (pleurothotonus): epidemiology and management. CNS Drugs 2002;16:165–74. Suzuki T, Kurita H, Hori T, Sasaki M, Baba A, Shiraishi H, et al. The Pisa syndrome (pleurothotonus) during antidepressant therapy. Biol Psychiatry 1997;41:234–6. Villarejo A, Camacho A, Garcia-Ramos R, Moreno T, Penas M, Juntas R, et al. Cholinergic– dopaminergic imbalance in Pisa syndrome. Clin Neuropharmacol 2003;26:119–21. Yassa R, Nastase C, Cvejic J, Laberge G. The Pisa syndrome (or pleurothotonus): prevalence in a psychogeriatric population. Biol Psychiatry 1991;29:942–5. Yohanan M, Aulakh JS, Weith J, Hawkins JW. Pisa syndrome in a patient in a wheelchair taking valproic acid. Am J Psychiatry 2006;163:325–6. Ziegenbein M, Schomerus G, Kropp S. Ziprasidone-induced Pisa syndrome after clozapine treatment. J Neuropsychiatry Clin Neurosci 2003;15:458–9.
Contents lists available at ScienceDirect
Progress in Neuro-Psychopharmacology & Biological Psychiatry j o u r n a l h o m e p a g e : w w w. e l s e v i e r. c o m / l o c a t e / p n p b p
Letter to the Editor (Case report)
Chronic Pisa syndrome associated with switching antipsychotics from olanzapine to ziprasidone Yi-Wei Yeh a, Yi-Chyan Chen a, Chih-Kang Chen a, Hui-Ming Feng b, Tzu-Yun Wang a, Chih-Lun Chen a,⁎ a b
Department of Psychiatry, Tri-Service General Hospital, National Defense Medical Center, Taipei, Taiwan Division of Medicine, National Defense Medical Center, Taipei, Taiwan
1. Introduction Pisa syndrome (PS), so called pleurothotonus, a rare asymmetric axial dystonia characterized by tonic lateral flexion of the trunk, has been mainly observed in patients using typical antipsychotics (Stubner et al., 2000; Suzuki and Matsuzaka, 2002). It also developed in rare cases using atypical antipsychotics and in patients with neurodegenerative disorder such as Alzheimer's disease or Parkinson's disease (Yassa et al., 1991; Suzuki and Matsuzaka, 2002). Ignoring PS will decrease patients' ability to participate in therapy and rehabilitation and will influence their quality of life. Here we present a patient with schizophrenia, who developed chronic PS for 2 years after switching antipsychotics from olanzapine to ziprasidone; the dystonia vanished after the cessation of ziprasidone.
2. Case report A 38-year-old single unemployed male had a history of paranoid type schizophrenia (meeting DSM-IV criteria) for 15 years. He had no history of perinatal or developmental abnormality, general medical disease or drug abuse. From 1993 to 2005, three different antipsychotics were used successively for 4–5 years each, including methylperidol 20 mg/day, sulpiride 600 mg/day, and olanzapine 10–20 mg/day. Weight gain of 25 kg, increase in waist circumference by 18 cm, and dyslipidemia were found during 2002–2005 under olanzapine treatment. To avoiding worsening of the metabolic syndrome, the antipsychotic was switched to ziprasidone 160 mg/day in July 2005 in acute ward and maintained in day-care ward during 2005–2007. However, one month after using ziprasidone, the patient began leaning to one side. This change progressed to the point where he was tilted at an angle of 25–30°. The leaning posture was persistent and refractory to anticholinergic (trihexyphenidyl 5 mg/day) administrated under the initial impression of tardive dystonia. The patient seemed unaware of and unconcerned about this change. He did not receive any change on his medication in day hospitalization until August 2007 when he was admitted to our hospital because of a psychotic episode. A posture of tonic flexion of his trunk and tilting his
⁎ Corresponding author. Department of Psychiatry, Tri-Service General Hospital, No. 325, Sec. 2, Cheng-Kung Road, Nei-Hu Area, Taipei 114, Taiwan. Tel.: +11 886 2 26739211; fax: +11 886 2 87927221. E-mail address: [email protected] (C.-L. Chen). 0278-5846/$ – see front matter © 2008 Elsevier Inc. All rights reserved. doi:10.1016/j.pnpbp.2008.11.001
head to the right side while sitting, standing, and walking was observed. He showed no other signs or symptoms suggestive of other EPS. The differential diagnoses of hysterical and catatonic presentation were both ruled out. Brain CT and EMG did not reveal any abnormality. Pisa syndrome was diagnosed. After switching to amisulpride 600 mg/ day for one week, the dystonia resolved completely without adjuvant treatment. Two weeks later, his psychotic symptoms also improved (CGI-S: 4–5, CGI-I: 2–3). At 12 months follow-up, the patient did not manifest any motor complication. 3. Discussion Ziprasidone, a serotonin-dopamine antagonist, has higher 5-HT2A/ D2 ration among atypical antipsychotics, and it is suggested that it has a lower risk of EPS (Schmidt et al., 2001; Dayalu and Chou, 2008). However, Pisa syndrome (PS) is a rare involuntary movement disorder and is distinguishable from other EPS. First, PS occurs mostly during the first month of treatment with atypical antipsychotics, whereas acute dystonia occurs within the first 24 h of treatment (Dayalu and Chou, 2008). Only 40% of reported PS responded to treatment by anticholinergics (Suzuki and Matsuzaka, 2002); its response is better than tardive dystonia but inferior to acute dystonia. PS mostly vanished after the discontinuation or reduction of the offending drug without any adjunctive treatment (Stubner et al., 2000), but tardive dystonia might persist (Dayalu and Chou, 2008). Consistent with previous reports, PS developed one month after using ziprasidone and persisted for 2 years under maintenance treatment; it resolved entirely upon discontinuation of ziprasidone even after a two-year course. Such a cause-and-effect relationship between the use of ziprasidone and the appearance of PS implied ziprasidone-induced PS (the Naranjo causality-scale score was 6). Although most evidence supports the possible pathophysiology of dopaminergic–cholinergic imbalance in development of Pisa syndrome (Stubner et al., 2000; Suzuki and Matsuzaka, 2002; Villarejo et al., 2003; Duggal et al., 2004), a serotonin–norepinephrine effect as well as dopamine interaction as causes of PS development should nevertheless be taken into account (Remington, 1988; Suzuki et al., 1997; Suzuki and Matsuzaka, 2002), particularly for ziprasidone with its rich pharmacologic profile of moderate serotonin–norepinephrine reuptake inhibitor, 5-HT1A agonism, and 5-HT1B/D, 5-HT2A/2C antagonism (Schmidt et al., 2001). However, the possible causality of PS might be specific to the introduction of a new drug (Suzuki and Matsuzaka, 2002), the
Letter to the Editor (Case report)
discontinuation of a former drug (Stubner et al., 2000), or the process of drug switching (Duggal et al., 2004; Rota et al., 2007). To date, only two cases of ziprasidone-associated PS have been reported. Ziegenbein et al. (2003) reported that a 38-year-old schizophrenic woman developed PS on day 18 while switching from clozapine 275 mg/day to ziprasidone 80 mg/day. The PS disappeared after withdrawal of ziprasidone and switching to amisulpride 14 days later. Duggal (2008) described a 20-year-old man with schizoaffective disorder comorbid with cocaine dependence. The patient showed acute pharyngolaryngeal dystonia and PS after administration of ziprasidone 120 mg/ day and valproate 1000 mg/day on day 2. The dystonia resolved after addition of anticholinergics. The author did not indicate the psychotropic medication prior to ziprasidone and could not exclude the possible effect of valproate (Yohanan et al., 2006; Duggal, 2008). Coincidentally, our case is similar to that reported by Ziegenbein et al. (2003), in that PS developed during atypical antipsychotic treatment after switching from one drug to another. In contrast to ziprasidone with its weak anticholinergic properties, both clozapine and olanzapine have potent anticholinergic effects. Switching antipsychotics with different anticholingeric effects could increase the risk of a cholinergic rebound (Lambert, 2007) and might result in dopaminergic-cholinergic imbalance. From this point of view, switching antipsychotics maybe a potential risk factor for development of PS (Duggal et al., 2004; Rota et al., 2007) should be performed slowly and gradually, especially when the former antipsychotic has a potent anticholinergic effect. 4. Conclusion Clinicians and patients easily neglected the PS. At least one study had shown that only 25% patients with schizophrenia are aware of their involuntary movements (Caracci et al., 1990) and that this was
163
correlated with their insight level. Clinicians have to keep in mind this motor complication to avoid worsening of symptoms and resulting in iatrogenic accidents. References Caracci G, Mukherjee S, Roth SD, Decina P. Subjective awareness of abnormal involuntary movements in chronic schizophrenic patients. Am J Psychiatry 1990;147:295–8. Dayalu P, Chou KL. Antipsychotic-induced extrapyramidal symptoms and their management. Expert Opin Pharmacother 2008;9:1451–62. Duggal HS. Acute Pisa syndrome and pharnygolaryngeal dystonia due to ziprasidone. J Neuropsychiatry Clin Neurosci 2008;20:108–9. Duggal HS, Sivamony S, Umapathy C. Pisa syndrome and atypical antipsychotics. Am J Psychiatry 2004;161:373. Lambert TJ. Switching antipsychotic therapy: what to expect and clinical strategies for improving therapeutic outcomes. J Clin Psychiatry 2007;68(Suppl 6):10–3. Remington GJ. The Pisa syndrome: possible role for serotonin and noradrenaline. J Clin Psychopharmacol 1988;8:228–9. Rota E, Bergesio G, Dettoni E, Demicheli CM. Pisa syndrome during aripiprazole treatment: a case report. Prog Neuropsychopharmacol Biol Psychiatry 2007;31:286–7. Schmidt AW, Lebel LA, Howard Jr HR, Zorn SH. Ziprasidone: a novel antipsychotic agent with a unique human receptor binding profile. Eur J Pharmacol 2001;425:197–201. Stubner S, Padberg F, Grohmann R, Hampel H, Hollweg M, Hippius H, et al. Pisa syndrome (pleurothotonus): report of a multicenter drug safety surveillance project. J Clin Psychiatry 2000;61:569–74. Suzuki T, Matsuzaka H. Drug-induced Pisa syndrome (pleurothotonus): epidemiology and management. CNS Drugs 2002;16:165–74. Suzuki T, Kurita H, Hori T, Sasaki M, Baba A, Shiraishi H, et al. The Pisa syndrome (pleurothotonus) during antidepressant therapy. Biol Psychiatry 1997;41:234–6. Villarejo A, Camacho A, Garcia-Ramos R, Moreno T, Penas M, Juntas R, et al. Cholinergic– dopaminergic imbalance in Pisa syndrome. Clin Neuropharmacol 2003;26:119–21. Yassa R, Nastase C, Cvejic J, Laberge G. The Pisa syndrome (or pleurothotonus): prevalence in a psychogeriatric population. Biol Psychiatry 1991;29:942–5. Yohanan M, Aulakh JS, Weith J, Hawkins JW. Pisa syndrome in a patient in a wheelchair taking valproic acid. Am J Psychiatry 2006;163:325–6. Ziegenbein M, Schomerus G, Kropp S. Ziprasidone-induced Pisa syndrome after clozapine treatment. J Neuropsychiatry Clin Neurosci 2003;15:458–9.