Abstract
K. Jagadheesan, and S. Haque Nizamie, Central Institute of Psychiatry, Kanke (PO), Ranchi, India:
Pisa syndrome is a rare dystonic reaction that occurs during treatment with typical antipsychotics [1]. Recently, this adversity has been noted in association with atypical antipsychotics such as clozapine [2], sertindole [3] and olanzapine [4]. Although tardive dystonia has been noted [5], presently, Pisa syndrome, comprising all its features, during risperidone therapy has not been documented in the literature. Herein we present a case of risperidone–induced Pisa syndrome.
A 24-year-old female with mental retardation, with a family history of mental retardation (maternal cousin), was brought with an illness of 10 years duration being characterised by inappropriate laughter, aggression, suspiciousness and disorganised behaviour. Her first mental status examination revealed irritable affect, delusions of persecution and reference and second person auditory hallucinations. Except poorly developed secondary sexual characteristics, she had no other physical abnormality. With the diagnosis of mild mental retardation and unspecified psychosis, she was started on risperidone 2-mg/day and trihexyphenidyl 2-mg/day. After a fortnight, she developed symptoms which included tilting of her body towards backwards and to the left side and tremors and cogwheel rigidity of the extremities. Hence, trihexyphenidyl was increased to 4–6-mg/day. Although her psychosis responded to risperidone, because of the persisting truncal tilting, it was discontinued and olanzapine 5-mg/day was started. After 2 weeks of olanzapine therapy, since there was some improvement in her motor abnormality and absence of psychotic symptoms, trihexyphenidyl was discontinued. However, a fortnight later her truncal tilting worsened and she was restarted on trihexyphenidyl (4–6-mg/day). Her truncal abnormality did not improve even after 4 weeks of trihexyphenidyl addition and subsequently she was lost to follow-up.
Pisa syndrome, an axial dystonia with flexion of the trunk towards one side, appeared for the first time during risperidone therapy in this patient who had no exposure to typical antipsychotics. Also, these symptoms became worse during olanzapine therapy following discontinuation of trihexyphenidyl. Notably, Pisa syndrome that started with smaller doses of risperidone, which also persisted during olanzapine therapy, did not improve with trihexyphenidyl. Similar to our patient, in an earlier study [6], a significant proportion of patients of Pisa syndrome had no improvement with anticholinergics.
Putative risk factors of Pisa syndrome include previous treatment with classical antipsychotics, combined pharmacological treatment, female gender, old age and the presence of an organic brain disorder [4]. In addition to the female gender, our patient also had mental retardation. The fact that Pisa syndrome of our patient did not improve during treatment and consequently, patient was lost to follow-up suggest that the onset of Pisa syndrome may cause discontinuation of the responsible pharmacological agents in spite of their therapeutic benefits. Thus, we suggest cautious use of risperidone and olanzapine in the patients who are prone to this distressing syndrome.