We report a case of 20-year-old male, belonging to lower socioeconomic class, educated up to 2nd standard, presented with severe unilateral dystonic left sided neck movements [Figure 1]. Careful history exploration revealed he was taking risperidone 2 mg irregularly for 2 months and then olanzapine 5 mg for another 4 months.
At 19 years, the patient presented with occasional anger outbursts, getting provoked on small matters and beating family members, running away from home, screaming episodes occasionally, fearfulness, sleep disturbance for 2 days; which was precipitated after fever. According to the mother, one friend might have threatened/made fun of him actually and after that patient stopped going out of house, and displayed above mentioned symptoms. This was interpreted as psychosis with persecutory ideas, and he was treated with risperidone 2 mg/day for 2 months and then with olanzapine 5 mg/day for 4 months.
In last two follow-ups patient did not present himself, and mother reported unusual neck movements, which were taken as a part of his overall psychopathology and not taken seriously, slight intermittent neck movements reported were missed as part of adolescent behavior problems mimicking some hero in movies.
As neck dystonia increased, the patient had a severe disability as patient had to keep his hands behind his head for the support. The movement would decrease when the patient was lying down and was absent during sleep. He stopped going outside or doing household work. Sitting comfortably or walking straight became impossible in waking hours. He even stopped taking food due to severe neck movements making chewing and swallowing difficult. His Abnormal Involuntary Movement Scale scoring was in severe rating.
His birth and early developmental milestones were normal. During 2-10 years of age patient was inattentive and mildly hyperactive. Other siblings were educated with Master's degree, and patient was also sent to school, but due to inattention and restlessness, he did not pass 2nd standard after three attempts. He left the schooling. With average executive functioning and life skills, he worked as an unskilled laborer in the neighborhood shops as a helping hand. Nobody suspected him of having an intellectual disability. Between 11 and 18 years, he was found to be getting over familiar, cheerful, moody, and short tempered. Sometimes, the patient had inappropriate social judgment; for which his friends made fun of him, and teased him.
On mental status assessment, patient was found to be having borderline intellectual functioning with standard intelligence quotient test. Routine investigations, thyroid function tests, electroencephalogram, fundus examination, cervical X-ray, magnetic resonance imaging brain were normal. After consulting neurophysician, Wilson's disease and other secondary causes of dystonia were ruled out.
The patient was treated with clonazepam 1 mg total dissolved solid (TDS), tetrabenazine 25 mg TDS, trihexiphenidyl 2 mg bipolar disorder (BD). After 2 months, there was some improvement of around 30%. Baclofen 10 mg was added; increased up to 20 mg, trihexyphenidyl reduced to 2 mg. With little improvement after 4 months of treatment for dystonia, levodopa + carbidopa (100 + 25) was added by neurophysician and increased up to 1/2 tablet TDS and baclofen omitted.
After 12 months of treatment, patient has improved around 90% with tetrabenazine 75 mg, levodopa + carbidopa (100 + 25) - 1/2 tablet BD, and clonazepam 1 mg BD. He has shown no psychotic or behavioral symptoms and is not on any antipsychotics.