Psychotic disorders in children are not common, however it is generally agreed that its prevalence increases during adolescence. The prevalence of psychosis in 13 to 19-year-olds was reported to be 0.54%, increasing from 0.9 per 10 000 at age 13 years to 17.6 per 10 000 at 18 years.8 Among adults with schizophrenia, 5% of them report onset of psychosis before the age of 15 years old and 20% of them have their onset before the age of 20 years old.9,10 Nevertheless, psychotic disorders in adolescence often mark the beginning of a lifetime contact with mental health services.
Psychotic disorders in adolescence can pose not only diagnostic and treatment challenges, but also difficulties to the adolescent as well as their families. As can be seen in MLS’s case, there were challenges since the initial onset of symptoms including diagnostic uncertainties, difficulties faced by MLS and his parents due to the uncertainty in diagnosis which then led to worsening of symptoms and a rehospitalisation. In addition, the onset of symptoms at the age of 17 would be of significance for MLS as this is the age where several important life events would take place such as the major school examinations Sijil Pelajaran Malaysia, which may determine his academic and occupational future, in addition to other important life transitions such as having a relationship with the opposite sex, completing school and making a decision regarding the next step in his life such as whether or not to pursue further education. As such, accurate and early diagnosis, with appropriate treatment is of utmost importance to ensure achievement of remission and to minimize any disabilities.
Though schizophrenia in prepubertal children is rare, the prevalence of schizophrenia in adolescents is approximately 1 to 2 per 1,000, with an estimated ratio of 1.67 boys to 1 girl. The rate of onset is reported to increase during adolescence, and while the onset of illness is usually insidious, it may occur suddenly in a previously well child, such as in MLS’s case. Multiple aetiological factors contribute to the development of schizophrenia, including genetic, neurodevelopmental, as well as environmental risk factors.11 There is a family history of psychiatric illness among 2 of MLS’s paternal relatives and although their diagnoses could not be ascertained, it is likely that his paternal uncle had suffered from a psychotic illness in the past from the description given. This contributes to genetic loading which poses as a risk factor for the development of MLS’s illness.
Another significant finding is that child and adolescent-onset schizophrenia is associated with premorbid developmental and social impairments. Some children and adolescents with schizophrenia are premorbidly more likely to have lower intelligence quotient, social withdrawal, isolation, poor peer relationships, excessive anxiety and academic trouble as compared to adult-onset schizophrenia, while some others may have histories of delayed motor or language milestones similar to some symptoms of autistic disorder. 12,13 A study on child and adolescent-onset psychoses found significant difficulties in social development affecting ability to make and keep friends in a third of cases with schizophrenia.14 MLS had poor eye contact as a child and although he did not have any other features of autism, he appears to be more socially reserved, having only few friends. This puts him at risk of developing schizophrenia, however whether premorbid impairments pose as a risk or precursor of psychosis remains a question. These premorbid impairments may be a causative factor for psychosis, or on the other hand, could be markers of an underlying neuropathological process which may be the cause of both the premorbid social impairment and psychosis.
In addition, MLS had reported seeing shadows on a few occasions during childhood, at age 10 and 12 years old. An association between self-reported psychotic symptoms during childhood and later schizophrenia has been found.4 A 15-year longitudinal cohort study demonstrated a very high risk of schizophreniform disorder at age 26 among individuals who reported psychotic symptoms at the age of 11 with an odds ratio of 16. 42% of the schizophreniform cases at age 26 reported one or more psychotic symptoms at age 11 years, including hallucinatory experiences and delusional beliefs. The individuals who reported the psychotic symptoms at age 11 did not have mania or depression at age 26, suggesting specificity of prediction to schizophreniform disorder.15 Though this study found an association between psychotic symptoms in childhood and later schizophreniform disorder but not schizophrenia, it appeared that attenuated psychotic symptoms contribute a significant high-risk premorbid phenotype.4
Growing up, MLS’s parents executed different parenting styles towards him. While his father gave him freedom and did not seem very involved in his daily living, his mother was overinvolved and controlling of his activities. The different types of parenting were described by Diana Baumrind in 1966 who introduced three models of parental control, namely authoritarian, authoritative and permissive. In permissive parenting, the parent is non-punitive and acceptant towards the child’s actions and desires, makes few demands for responsibility, allows the child to regulate his own activities and avoids the exercise of control.16 The parent imposes few maturity demands and either indulges or neglects the child’s needs.17 On the other hand, authoritarian parenting is characterized by high expectations of conformity to parental rules. The parent is obedience-oriented and the child is expected to obey orders without explanation. According to Baumrind, both permissive and authoritarian type of parenting may prevent the child from being able to engage in interaction with people. Children and adolescents of authoritarian parents are tend to have less self-confidence and become socially withdraw, while permissive parents tend to suffer from problems with emotional regulation and self-control, and thus they were reported to be more likely involved in problematic behaviours and aggression.18 In MLS’s case, as MLS’s mother plays a more dominant role in the family compared to his father, her authoritarian parenting had resulted in MLS becoming more socially withdrawn, mostly staying at home with few friends. More importantly, this had resulted in a lack of bonding between MLS’s mother and himself.
MLS had started to develop symptoms of unusual behaviour 3 days after being discharged from Klang Hospital for dengue fever, in addition to being socially withdrawn and deterioration in functioning. Though MLS seemed preoccupied when admitted to University Malaya Medical Centre the first time, he was unable to describe any positive symptoms of psychosis until the second admission when he had developed auditory hallucinations, persecutory delusion and thought echo. It has been reported that children and adolescents with psychosis can typically present with a prodromal period characterized by deterioration in personal functioning and negative symptoms such as concentration and memory problems, unusual behaviour, bizarre perceptual experiences, social withdrawal, apathy and reduced interest in daily activities, some of which are observed in MLS’s case. These symptoms may follow an acute period of stress or a physical illness, which was dengue fever in MLS’s case.19 Frank psychosis develops within 12 months of symptom onset in 40% of patients20, and this prolonged duration of prodromal period may affect school performance as well as delay the diagnosis of psychosis or schizophrenia, but for MLS the psychotic symptoms developed much earlier.
The acute episode which follows the prodromal period is usually characterize by positive symptoms of hallucinations and delusions, which may lead to a sense of fear of puzzlement during the period of delusional mood. As perceptual disturbances may be new experiences for children or adolescent, they may be distressed or confused, as can be observed in MLS who appeared confused and preoccupied, and may subsequently develop secondary delusional belief such as being replaced with doubles when they experience themselves or family members as being unfamiliar. As for MLS, the lumbar puncture performed on him in the neuromedical ward may have strengthened the persecutory delusion where he started to believe that someone intentionally removed a bone from his spine and further persecutory delusion such as the belief that people wanted to poison his food in the ward.
A detailed assessment including thorough history and physical examination as well as diagnostic workups need to be performed as psychosis in childhood and adolescence may result from an organic cause such as infection as well as neurological, autoimmune, endocrine or metabolic conditions. Neuropsychiatric conditions such as encephalitis, temporal lobe epilepsy, cerebral lupus, drug intoxication as well as neurodegenerative illnesses such as Wilson’s disease need to be ruled out particularly when there are neurological signs or fluctuating levels of consciousness, in which blood tests, computed tomography (CT) scan, MRI or electroencephalography (EEG) may be helpful.19 As for MLS, organic workup was performed during both the admissions to UMMC due to the acute onset of abnormal behaviour and presentation after dengue fever during the first admission, as well as an episode of recorded fever and confusion during the second admission.
MLS was investigated for post-dengue encephalitis during the first admission to UMMC. Dengue viral infection can present with varying clinical manifestations ranging from asymptomatic infection to life threatening haemorrhagic fever and dengue shock syndrome. Apart from the cmp renal and hepatic dysfunction, dengue fever can present with neurological complications including dengue encephalopathy, encephalitis, neuromuscular complications as well as neuro-ophthalmic involvement in 4-5% of confirmed dengue cases.21 While the more common symptoms of encephalitis include headache, disorientation and seizures, neuropsychiatric symptoms such as psychosis, mania and dementia have been reported as post-infectious sequelae.22 A case report in 2013 described a 12-year old boy who presented with behavioural change characterized by emotional lability, dependence, alteration in rhythm of language and tone voice, anxiety and change in tastes in addition to fever, severe headache, seizures and vomiting. Though magnetic resonance and computed tomography showed no brain changes, serology for dengue was positive for both IgM and IgG and cerebrospinal fluid revealed high protein levels and increased lymphocytes. The boy had improved with risperidone, but behavioural symptoms were still reported to be present 60 days later. 23 Another author reported a 21-year old man who developed an episode of classical manic symptoms including overactivity, excessive talking, argumentativeness, irritability, grandiosity, abusiveness and decreased need for sleep on the 6th day of dengue illness with thrombocytopenia, and was successfully treated with carbamazepine and haloperidol.24 The diagnosis of dengue encephalitis may be made either by detection of virus or antibodies in the cerebrospinal fluid, but the absence of antibodies will not rule out encephalitis. MRI, the modality of choice of brain imaging, may show findings consistent with viral encephalitis such as cerebral oedema, white matter changes, brain atrophy and necrosis.25 In MLS’s case, there was no evidence suggestive of infection in his blood or cerebrospinal fluid tests, while blood imaging and electroencephalography did not reveal any significant findings. This points against the diagnosis of post-dengue encephalitis, although the investigations done by the neuromedical team during both admissions were justified and thorough.
The management of adolescent psychotic disorder encompasses many aspects and should take into consideration a risk assessment to self and others, mental state, insight into illness, likely adherence to treatment as well as level of support available, in addition to any predisposing, precipitating, maintaining and protective factors. As children and adolescents with psychosis or schizophrenia and their families may experience significant distress, it is important to engage both the young person and their parents or carers in the management of their illness, which is the foundation of subsequent pharmacological and psychosocial interventions. The Early Pyschosis Declaration emphasizes the reduction of long delays that families face by services working better together and earlier to meet needs of young people and their parents. Among the issues that need to be considered in the management of children and adolescents with psychotic disorder include the normal developmental tasks of adolescents, effectiveness and safety of particular treatments as well as offering service and information to parents about costs and benefits of any recommended treatment. Psychoeducation for the young person and their families is important, such as in MLS’s case as his parents have limited awareness of mental illness and had missed MLS’s initial psychiatric appointment as they thought that he was well when he was still preoccupied and had started to develop positive psychotic symptoms. Explanation about the illness, guidance as well as involving family members in treatment decisions are important in ensuring compliance and continuity of care particularly as the young person such as MLS transits from adolescence to early adulthood.19
Though it is recommended that psychosocial and other benign options should be employed before considering medication, pharmacological treatment such as antipsychotics has been more widely used in recent times. Though there are few benefits of second generation antipsychotics over first generation antipsychotics in efficacy in treating early-onset schizophrenia and schizoaffective disorder, the former is less likely to cause side effects of extrapyramidal symptoms but at the same time are associated with weight gain, metabolic problems and risk of diabetes, as part of the findings from the treatment of early-onset schizophrenia spectrum disorders study (TEOSS).26 As such, it has been recommended that lifestyle and dietary advise should be given in addition to side effect monitoring when initiating an antipsychotic medication.27
Though antipsychotic medication is effective in reducing positive psychotic symptoms, with a modest effect size of 0.2 to 0.3, there is limited evidence for antipsychotic treatment of psychosis and schizophrenia in young people, with only minimal differences in efficacy found among the different antipsychotics but with large differences in side effect profiles.28 A Cochrane review of antipsychotic medication used in children below age 13 years with childhood-onset schizophrenia found inconclusive evidence regarding the effects of antipsychotic medication for early-onset schizophrenia, with clozapine showing benefits over haloperidol in treatment resistant schizophrenia but were offset by risk of serious adverse effects.29 Another meta-analysis on the efficacy, safety and tolerability of antipsychotics in adolescents aged 13 to 17 years with schizophrenia demonstrated that antipsychotic treatment with risperidone, olanzapine or aripiprazole resulted in significant improvement in symptomatology, and that treatment with 10mg daily dose of aripiprazole was associated with the lowest incidence of extrapyramidal symptoms with no significant weight gain.30 However, most antipsychotic medications have not been approved to treat early-onset schizophrenia as they have not been tested in the younger individuals. In Europe, aripiprazole and more recently paliperidone which was approved the use in adolescent schizophrenia, while aripiprazole, olanzapine, paliperidone, risperidone and quetiapine are approved by the Food and Drug Administration (FDA) in the United States for the treatment of early-onset schizophrenia in individuals aged 13 to 17 years old.31 Antipsychotic treatment was started for MLS due to the severity of the psychotic symptoms as well as the risk of harm to self and others. As he had developed side effects of sedation with a low dose of risperidone, it was changed to aripiprazole, which was then switched to olanzapine when he did not respond to aripiprazole at a dose of 10mg daily. His symptoms improved with olanzapine and had maintained well with this medication, but he would need to be regularly monitored for weight gain and metabolic side effects. As he had started to experience weight gain, dietary advice was given and continuation of active lifestyle and exercise which he was beginning to engage in was encouraged.
Psychological interventions such as cognitive behavioural therapy have been studied and found that cognitive behavioural therapy can be possibly used to reduce the strength of delusional beliefs as well as to reduce the likelihood of relapse among young individuals with first episode of psychosis. In addition cognitive remediation therapy, art therapy as well as structured employment approaches are being developed and evaluated.19 Family intervention adapted for the developmental needs of adolescents aiming to reduce criticism and hostility towards the adolescent may also be helpful.31 In MLS’s case, a lesser overinvolvement by his mother was encouraged as overprotection and emotional overinvolvement are associated with high expressed emotion, which are in turn associated with a high relapse rate among patients with schizophrenia.
More importantly for MLS is the return of functioning and continuation of schooling as this is his final year of secondary education and also the year in which he is bound to sit for the Sijil Pelajaran Malaysia examinations. It may be a stressful situation for MLS going back to school after a duration of 2 months of not attending school, in addition to the stress of catching up with studies, preparing for exams as well as possible stigma from others in school. It is important for school staff to support young individuals with psychosis or schizophrenia in school as they may feel distressed by their psychotic symptoms which may be worsened by responses of those around them, particularly if they get mocked or bullied by others. In addition, exposure to environments or classes with high levels of expressed emotion should be minimised as they are known to increase the risk of relapse but at the same time provide opportunities for social interaction with others.19
As there is a risk of future relapse with ongoing stressors such as sitting for examinations, expanding academic and career capabilities as well as undergoing the transition from adolescence to adulthood, ongoing follow-up is recommended for MLS. Among the factors contributing to a poorer prognosis in child and adolescent-onset psychosis include premorbid social and cognitive impairments, prolonged first psychotic episode, prolonged duration of untreated psychosis and negative symptoms. Schizophrenia in young individuals typically run a chronic course, with only 12% found to be in full remission at discharge and those with full recovery most likely recover within first 3 months of onset of psychosis.19 Nevertheless young individuals who do not undergo complete recovery from the illness may still be able to maintain an acceptable quality of life with adequate support as recovery is a personal process involving finding a sense of self and hope. As for MLS, regular follow-up and assessment of treatment needs in addition to return to functioning, prevention of a future relapse as well as continuous support from family members are crucial to ensure a good quality of life as an adolescent and a smooth transition to adulthood.