Abstract
Catatonia is uncommon in children and adolescents. Cohen et al. reported 42 cases of catatonia in children and adolescents [1]. Only three patients were under 13 years of age. Two of these were treated with and responded to electroconvulsive therapy (ECT). Of the total of 42 patients, 22 had been treated with ECT.
Cizadlo and Wheaton reported on an 8½-year-old girl with major depressive episode with catatonic symptoms who responded to ECT [2]. Fink and Carlson reported that ECT was rarely used in children and adolescents in spite of favourable treatment outcomes for depression and catatonia in prepubertal children [3].
Case report
An 11-year-old girl was medically evacuated from a provincial town to a tertiary child psychiatric inpatient unit after 3 days of unusual behaviour. She had no past medical or psychiatric history.
Three nights prior to presentation at her local hospital she ran through the house turning on all the lights. She experienced nihilistic auditory hallucinations about the world ending and feared for the safety of her family. On one occasion she commented that her mother was dead.
At the provincial hospital's emergency department she reported experiencing command hallucinations to kill people. For 3 days she refused all fluids and food intake for fear of poisoning by staff.
She was treated with i.v. haloperidol 0.5 mg as needed (PRN) and received 2 mg in total over 90 min. She developed dystonic reactions at this dosage. She subsequently received i.m. olanzapine 2.5 mg up to a total of 30 mg over 2 days. She also received i.v. midazolam 2.5 mg interspersed with haloperidol and olanzapine with little effect. This regimen of medication offered brief respite from her agitation. She required 3–4 members of staff to continuously restrain her to prevent injury or escape.
Computed tomography brain scan was found to be normal. Due to the urgency of the presentation she was medically evacuated by aeroplane under midazolam infusion to a tertiary paediatric teaching hospital.
The patient's mother reported that the girl had been uncharacteristically oppositional and defiant in the preceding weeks and she became increasingly withdrawn from her peers. For 2 days prior to her hospitalization she had shown strange facial grimacing and assumed bizarre postures. She also had increased motor activity. When she was not posturing she would throw herself onto the ground.
The only identifiable stressor was parental separation 4 months previously. Her father's departure had caused the patient significant grief. One month after the separation the patient's sleep deteriorated. Despite not sleeping well she continued to be involved in multiple sporting activities.
Premorbidly, the patient was a talented athlete. She was a high achiever at school and socialized well with peers.
There was a family history of mental illness: her paternal grandmother had schizophrenia and her maternal grandmother and maternal uncle had bipolar disorder. Her father had a history of alcohol abuse and depression.
On presentation to the paediatric hospital the patient was disorientated. At one stage she did not recognize her mother. She was admitted to the child psychiatry unit. She presented with the catatonic symptoms of extreme negativism, excessive motor activities and verbigeration. The patient initially refused to eat or drink although she did manage minimal fluid and food intake 2 days into her admission. She refused oral medication.
Investigations were undertaken to exclude organic causes. Magnetic resonance imaging showed no abnormality. Lumbar puncture was unremarkable. Serology for cytomegalovirus and Epstein–Barr virus showed past exposure. Hepatitis B and C, Herpes simplex virus 1 and 2, parvo virus, Toxoplasmosis gondii and syphilis serology were negative. Human chorionic gonadotropin was also negative. Plasma amino acids level was normal. Lead and mercury levels were normal. There was no evidence of illicit drugs on urine screen and no urinary tract infection. Thyroid function, liver function and electrolytes, glucose and renal function were normal.
The patient was treated with i.m. olanzapine up to 10 mg day–1 for 5 days with no response. The olanzapine was increased to 20 mg day–1 with little effect. Diazepam 15 mg day–1 was added to the olanzapine for sedation and the patient's confusion, disorientation and agitation worsened.
Despite attempts to contain her agitation with nursing care and i.m. medications, she continued to display psychotic phenomena and exhibit dangerous behaviour. She feared that she would be killed and requested staff kill her. She expressed nihilistic delusions that she was dead. She repeatedly attempted to abscond from the unit and continued to physically attack nurses in response to her paranoid beliefs. She engaged in serious deliberate self-harm with suicidal intent including scratching her neck resulting in deep excoriations, blocking her airway with tissue paper, banging her head against a concrete wall and attempting to cut her throat using glass from a light she broke. She was also observed to be displaying sexualized behaviours including masturbation and touching other people in a sexualized manner.
Due to the severity of her psychotic symptoms and self-injurious behaviour and the lack of response to psychotropic medication after 5 days, a second opinion for management with ECT was sought and obtained from another child and adolescent psychiatrist. Her parents consented to the procedure.
Bilateral ECT was performed and adequate seizure achieved at first treatment and she showed immediate improvement. There was decreased psychomotor agitation, she denied further suicidal thoughts and became more interactive. She continued to be agitated at times but responded to directions by the nursing staff.
She continued to improve on subsequent bilateral ECT. After six ECT treatments her mood was notably hypomanic. ECT treatments were stopped and she was commenced on sodium valproate 200 mg mane and 500 mg nocte and risperidone 2 mg nocte. Risperidone was gradually decreased to 0.5 mg nocte at discharge. After 7 weeks of hospitalization she was discharged with her psychotic and mood symptoms resolved completely.
Communication with the patient 1 year later indicated that she had returned to her baseline functioning. She returned full time at school and her academic performance continued to be of a high standard. She has returned to her state representative level sporting activity and she has returned to age-appropriate social functioning. She is no longer on psychotropic medication.
Discussion
This case highlights the difficulty in diagnosing and managing a prepubertal girl who presented with life-threatening symptoms of catatonia. There is a paucity of evidence for the use of ECT in this age group [1], [2].
Diagnosis of catatonia in a prepubertal child is difficult due to its rarity and lack of diagnostic uniformity. Two or more signs are required to make the diagnosis of catatonia [4–6]. The patient showed strange facial grimacing, assumed bizarre postures, increased motor activity, extreme negativism, excessive motor activities and verbigeration, which support the diagnosis of catatonia.
Medically treatable causes of catatonia were excluded. Initial metabolic and toxicological investigations ruled out organic causes. The patient initially presented with symptoms of delirium. In retrospect this was likely due to high doses of benzodiazepine and neuroleptic medications used to sedate her.
After the sixth ECT treatment she exhibited symptoms of hypomania. The hypomania did not subside with cessation of ECT. The elevation in mood responded to mood stabilization with sodium valproate. Risperidone was gradually decreased and she was discharged on 0.5 mg nocte.
We considered the diagnosis of malignant catatonia/neuroleptic malignant syndrome but the lack of pyrexia and the normal creatinine phosphokinase level excluded these differential diagnoses.
This case raised the ethical dilemma of using ECT in a child [7]. Due to the severity and relentless nature of her self-harming behaviours that were unresponsive to conventional medical treatment, ECT was considered and a second opinion was sought [6].
This case highlights the need for thorough clinical investigation, to provide diagnostic certainty and exclude organic causes. Sharing the dilemma and treatment options with the parents and acquiring a second opinion were also imperative to the decision to treat with ECT. The favourable treatment response to ECT in this life-threatening presentation was reassuring for both the family and the treating team.