Treatment resistant non-catatonic mutism in schizophrenia responding to a combination of continuation electroconvulsive therapy and neuroleptics.

Non-catatonic mutism in schizophrenia has been described less frequently in literature. We describe the case of a young male who presented with non-catatonic mutism, secondary to first rank symptoms, which was refractory to adequate antipsychotic trials (quetiapine, risperidone, aripiprazole, ziprasidone, and trifluperazine) and responded to a combination of electroconvulsive therapy (ECT) and neuroleptics partially. However, when the ECT was continued in the continuation phase, the patient started speaking.

Advancement in psychopharmacology has contributed immensely in reducing the pessimism centered on the treatment of schizophrenia. But unfortunately despite good compliance and adequate treatment nearly 10-30% of patients show little or no response to antipsychotic medications, additionally 30% of patients have partial response to treatment, with mild to severe residual hallucinations or delusions.[1] This has always compelled the clinicians to look for additional or alternative treatment regimens which could benefit the unresponsive or partially responsive subjects. Since its first use, electroconvulsive therapy (ECT) is has been used in the treatment of schizophrenia. Several studies suggest that ECT helps augment the action of antipsychotics in acute[2] as well as in treatment-resistant schizophrenia.[34] However, over the years the use of ECT in the treatment of schizophrenia has declined, but it still remains a treatment option particularly in the management of medication resistant subjects.[5] Combination of ECT and antipsychotics is usually indicated in patients who fail to respond to single regimens of antipsychotic agents, clozapine, or ECT alone.[6]

Continuation ECT (C-ECT) refers to treatments spaced at interval of 1 week to 1 month, administered after a successful ECT course to reduce the risk of relapse. When it is continued beyond 6 months, to reduce the risk of recurrence of a new episode of illness it is termed as maintenance ECT.[7] C-ECT combined with maintenance-neuroleptic medication has been proposed to be associated with better treatment outcome, improvement in quality of life and functioning than either treatment alone.[8]

Mutism is usually considered to be a manifestation of catatonia in schizophrenia, but mutism due to other forms of psychopathology has also been described. In a report on schizophrenia in Kosrae, mutism has been described as a cultural variant of psychopathology, in which most patients used non-verbal methods of communication. In 19 out of 22 patients with schizophrenia selective mutism was seen in the initial phase, without other associated catatonic features which responded to treatment with neuroleptics.[9] In a case report, Basanth et al. described a case of non-catatonic mutism in a subject with paranoid schizophrenia who did not respond to three courses of ECT and clozapine in the dose of 450 mg/day.[10]

We describe a case of non-catatonic mutism, secondary to first rank symptoms, which was refractory to antipsychotic treatment alone and responded to a combination of C-ECT and neuroleptics.

CASE REPORT

Mr. A 28 years, who premorbidly had schizoid traits with no family history of mental illness, presented to the emergency with mutism and negativism. On further evaluation, physical examination did not reveal any other signs of catatonia and routine blood tests (hemogram, serum electrolytes, renal function tests, liver function tests, fasting blood sugar, and lipid profile), electrocardiogram, chest radiography, magnetic resonance imaging of brain, and electroencephalography were within normal limits. In view of the mutism and negativism, he was initially started on lorazepam 8 mg/day with which his symptoms did not improve, following which he was shifted to the psychiatry inpatient unit.

In the inpatient unit, exploration of history revealed that the patient had been ill for 9 years, with illness having an insidious onset, continuous and progressive course characterized by academic decline, social withdrawal, apathy, reduced self-care, non-specific anxiety for initial 3 years followed by delusion of persecution, delusion of reference, thought broadcast, auditory hallucinations of commanding type, three suicide attempts, mutism and refusal to take food which responded minimally to adequate trials of quetiapine, risperidone, aripiprazole, ziprasidone, and trifluperazine. For the last 2 years the patient had stopped speaking completely and would occasionally respond by gestures or writing. He would frequently refuse to eat for days together. With the available information a diagnosis of paranoid schizophrenia (as per Diagnostic and Statistical Manual, 4th Revision) was made. In view of lack of response to five antipsychotics in adequate doses for adequate duration, possibility of treatment resistant schizophrenia was also envisaged. At the time of admission to the inpatient unit he was totally non-communicative and mostly remained confined to his bed. Initially the option of clozapine was discussed with the family members, but the family members refused consent, following which he was started on haloperidol which was increased to 25 mg/day. Along with haloperidol he was started on ECT after the consent of family members.

Modified bilateral ECT was administered thrice a week (on Mondays, Wednesdays, and Saturdays). Atropine was used as a premedication, thiopental sodium was used for induction, and succinylcholine was used for muscle relaxation.

By 7th ECT the patient started to respond emotionally to the therapist, participated in some ward activities and communicated his beliefs through writing. During these interactions the patient reported that he has commanding auditory hallucinations which ask him not to speak at all and threaten him of untoward consequences if he did so. Further, he also expressed that as people around him knew about his thoughts, there was no need for him to speak. ECTs and haloperidol were continued and with a total of 15 ECTs over a period of 40 days. However, there was no further improvement in psychopathology. Following stoppage of ECT, aripiprazole 20 mg/day was added to haloperidol, but his clinical state worsened again to the previous level and the patient became totally unresponsive. Following this ECT was restarted and he was again given eight ECTs. In the meanwhile family consented for clozapine which was started and gradually increased to 225 mg/day with regular hematological monitoring and aripiprazole was stopped. There was improvement in his symptomatology to the level seen previously with ECT over the period of 4 weeks. Following this it was decided to continue clozapine 225 mg/day, haloperidol 20 mg/day, and continue the patient on maintenance ECT after discharge. He was given C-ECT – initially 1 per week for 1 month followed by 1 ECT every fortnight. While on C-ECT, his clinical status improved significantly and he started communicating by 5th C-ECT. Meanwhile on mental status examination he claimed that the hallucinations fluctuated from time to time and which were primarily responsible for his not talking to others. He also disclosed that he had attempted suicide in the past due to the hallucinatory voices.

DISCUSSION

Studies have shown that use of C-ECT/maintenance ECT in treatment of resistant schizophrenia can lead to improvement in psychopathology, prevent relapse, and recurrence.[1112] Most practice guidelines do not emphasize the use of continuation and maintenance ECT in schizophrenia.[1]

Few studies have described non-catatonic mutism in subjects with schizophrenia. The index case was symptomatic for quite sometime, before presenting with mutism, without other catatonic signs. His organic workup was negative and once the patient started speaking he described that the hallucinations were the reason for his mutism. All these point to the fact that the mutism could be attributed only to positive psychotic symptoms. A case report in the past also described a case of non-catatonic mutism, which did not respond to ECT.[10] The index case responded partially to a combination of ECT and haloperidol initially as he started interacting through non-verbal means. However, the response was lost when ECT was stopped. This encouraged us to consider continuation of ECT. We continued with a combination of continuation ECT, clozapine and haloperidol, which possibly led to the improvement.

Very few studies and case series have described non-catatonic mutism in schizophrenia. Our case highlights the fact that if a subject with schizophrenia presents with mutism, not responding to antipsychotic medications, ECT should be considered, and depending on the initial response continuation ECT can be given, as the response in these cases may be slow. The case also highlights the fact that ECT can be given safely with combination of clozapine and haloperidol.