Catatonia in a 10-year-old boy with early childhood neglect and disruptive behaviours in psychiatric residential treatment

Background

Catatonia is a rare and serious neuropsychiatric syndrome that is often misdiagnosed in children and adolescents. Catatonia can cause a 60-fold increase in premature death, including suicide, in paediatric patients, when compared with the general population of same sex and age, with a 95% CI, based on a study done by Cornic et al.1–3 One study has shown a 5% prevalence of catatonia in outpatient paediatric psychiatric patients and a 17% prevalence in youth with psychotic disorders.4 Catatonia in childhood and adolescence has been poorly investigated and is thought to be underdiagnosed.5 6 A literature review of catatonia cases from birth to age 18 done in August 2020 by this author showed 309 reported cases. When narrowed to ages 6–12, 114 cases emerged with two published cases below the age of 12. In the literature review, the ages of cases cited were typically between 13 and 18. Most of the reported cases were in adolescents with autism, psychotic disorders, mood disorders and/or neurological disorders. No cases were found in children with trauma histories in long-term psychiatric residential care.

Publishing a case report in youth with trauma histories is challenging for multiple reasons. Barriers include obtaining consent from guardians, assent from youth (who often have developmental delays), the fluid nature of treating youth in state custody and incomplete histories due to multiple placements. Despite these obstacles, clinicians should not be deterred from publishing unusual presentations of serious medical conditions like this case, as there is increasing evidence that childhood maltreatment in its variant forms can be a risk for catatonia in children and adolescents.7 The lack of research in traumatised youth living in psychiatric congregate care makes it difficult to ascertain the number of paediatric catatonia cases in which trauma plays a role.

This report was prepared following the CARE guidelines.8 In this case, a 10-year-old boy with intrauterine exposure to alcohol and multiple drugs and early childhood deprivation developed catatonia 24 hours after the administration of a first-time intramuscular injection of haloperidol 5 mg for treatment of acute agitation and aggression. He did not have a primary psychotic disorder, autism or a medical or neurological condition that would predispose him to catatonia. His primary psychiatric diagnoses were severe oppositional defiant disorder (ODD), nighttime enuresis and borderline intellectual functioning. This boy’s other medical problems included severe obesity and metabolic syndrome, not related to antipsychotic usage. The precipitant for catatonia was first-time exposure to a high potency neuroleptic. A case such as this one, of a 10-year-old child in residential care, with a 24-hour delayed catatonic response to intramuscular haloperidol, which is often used to treat agitation in children in psychiatric and emergency facilities, has never been reported.

Case

A 10-year-old biracial (Caucasian/black) man with borderline intellectual functioning (Fullscale Intellectual Quotient (FSIQ)=76, WISC-intravenous (Wechsler Intelligence Scale for Children, 4th Edition), 2016), severe disruptive behaviours, nighttime enuresis, morbid obesity and metabolic syndrome, developed catatonia in a psychiatric residential centre, approximately 24 hours after receiving a first-time intramuscular dose of haloperidol 5 mg for agitation and severe aggression. The boy’s perinatal history is significant for intrauterine exposure to alcohol, cannabis, cocaine and methamphetamine. He was exposed to prostitution, drug use and violence when he lived with his biological mother until the age of 3, when he was taken into state custody due to neglect and placed into the custody of his 80-year-old maternal great-grandmother, who lived in another state. His biological father was unknown; mother worked as an exotic dancer and was arrested several times for prostitution and drug possession. His early childhood developmental history was mostly unknown, as his guardian lacked knowledge of his birth history and early childhood developmental experiences other than what has been relayed above. The child’s state social worker lacked further knowledge of his early developmental and medical history and lacked any other significant historical information about the child from his birth state. Lack of communication across state lines and poor historical information is not uncommon when children enter state custody for abuse/neglect and cross state lines. The child was living with his maternal great-grandmother, who was in her late 80’s and raising him on her own when he entered long-term psychiatric residential treatment. No other family members, including the child’s maternal grandmother and several maternal aunts, agreed to raise him, when he was placed in state custody at age three. After he was removed from his biological mother’s custody, he had no known further contact with her. The mother’s parental rights were never fully terminated; maternal great-grandmother remained his permanent guardian. Maternal family history was significant for addiction and depression.

He entered psychiatric residential treatment due to great-grandmother’s inability to control his impulsive behaviours like excessive overeating (ie, he would eat two extralarge pizzas as daily snacks) and unprovoked aggression such as destruction of school property and attacks on school staff and extended family. Examples of his violent behaviours include him causing a wrist fracture in a teacher and a rib fracture in another. Great-grandmother voluntarily admitted him for long-term psychiatric residential treatment with involvement from the state’s Department of Human Resources (DHR); great-grandmother retained custody while the state’s DHR paid for the treatment through a boarding home agreement. On admission to the intensive psychiatric residential treatment centre, he was morbidly obese—63 inches, 250 pounds, body mass index (BMI)=44 kg/m². His catatonic reaction to haloperidol occurred 10 months after admission, when he was severely obese—64 inches, 203 pounds, BMI=36 kg/m².

This boy spent 10 months in psychiatric residential treatment prior to receiving his first-time dose of haloperidol 5 mg. He had no prior exposure to an antipsychotic. The haloperidol injection was given at 12:45 hour after he attacked staff and destroyed property in his classroom. The boy’s behavioural problems, which were triggered by a conflict with a peer, started at approximately 11:00 hour. Behavioural interventions were ineffective, so an oral prn of hydroxyzine 50 mg was given at 11:45 hour and again at 12:15 hour prior to the child receiving the haloperidol injection at 12:45 hour, which involved a physical restraint (physical hold by multiple staff; no mechanical restraint) lasting less than 5 min. He was not given an anticholinergic agent with the haloperidol injection because he had received two doses of hydroxyzine 1 hour prior to the injection. He showed no immediate adverse reactions or side effects from the haloperidol injection other than mild sedation. He slept without incident the evening after receiving the injection.

At 8:00 hour the next day, he walked to his classroom with normal motor tone but poor eye contact, minimal social interaction and sleepiness. This is a child whose was typically very social and extraverted. Throughout the morning, he struggled to stay awake and did not participate in classroom activities. Catatonic symptoms emerged at approximately noon and included stupor, negativism and mutism. In addition, he was drooling, had no eye contact, complete social withdrawal, poor muscle tone, psychomotor slowing, no response to verbal commands and lethargy. He sat in his classroom chair limply, awake, but unresponsive to verbal commands. He had no cogwheeling. He met the Diagnostic and Statistical Manual Fifth Edition (DSM-V) diagnostic criteria for catatonia as he had 3/12 symptoms required for the diagnosis.3 His vital signs were within normal limits. He was assessed by his psychiatrist at 12:05 hour and diagnosed with neuroleptic-induced catatonia (NIC). He was given lorazepam 1 mg intramuscular once at 12:20 hours and showed modest improvement in symptoms within 30 min; vital signs remained stable. A second dose of lorazepam 1 mg intramuscular was given at 14:20 hours; he awakened and showed continued improvement of catatonic symptoms.

He was placed on a one-to-one for constant supervision. The primary care physician on duty assessed him at 15:00 hours and saw motor rigidity so the child received diphenhydramine 50 mg intramuscular at 15:15 hours. Vital signs were monitored every 15 min for the next 48 hours and blood pressure was in the 150s/90s throughout the evening, with a heart rate in the 80s and 90s. He had no fever. Within 36 hours, he had normal vital signs and complete resolution of catatonic symptoms. Lab studies included a complete blood count, basic metabolic panel and liver profile, and were within normal limits. He stabilised entirely in the intensive psychiatric residential treatment programme and did not require hospitalisation. His psychiatrist added the adverse reaction of catatonia, secondary to haloperidol, to his medical chart.

One year after admission (2 months after the catatonic reaction), the boy’s behaviours improved to the point that the team deemed he was ready for a slow transition home to live with his great-grandmother. During one of his extended home visits with his great-grandmother, she decided to stop treatment and move him to another state, against medical advice. At that time, he was 66 inches tall and weighed 173 pounds, BMI=29 kg/m². He was healthier, no longer met criteria for metabolic syndrome and showed improved self-regulation. Much to the concern of the treatment team, his guardian disappeared with the child, so he was not returned to the facility to complete his transition. Without proper home and community supports in place, the chances of the boy’s continued improvement minimised.

Differential diagnosis

The treating psychiatrist diagnosed the child with NIC due to the sequence of events (ie, catatonic symptoms occurring within 24 hours of first-time exposure to a D2 receptor antagonist and his positive therapeutic response to lorazepam, withdrawal of antipsychotic and supportive care). NIC, a type of catanoia, is considered part of a severe extrapyramidal reaction to a neuroleptic that has catatonic features and in severe cases, can lead to neuroleptic malignant syndrome (NMS).5 NMS shares similar clinical symptoms to catatonia but also includes hyperthermia and autonomic instability. Catatonia in children is typically associated with underlying psychotic and mood disorders. This child lacked symptoms of depression, mania or psychosis. Rather, he was disruptive, impulsive, and aggressive consistent with ODD and early conduct disordered (CD) behaviours. Furthermore, neurological and other medical conditions such as seizure, autoimmune disorders, infection and toxidromes were considered. The child had no seizure history and completely normal laboratory studies. Autism was considered but he had typical communication and social skills that did not meet criteria for autism spectrum disorder. In addition, the child’s early childhood and intrauterine history were considered as possible predisposing factors towards catatonia. His perinatal exposure to drugs and alcohol was significant and his early childhood neglect and exposure to drugs may have predisposed him to a catatonic reaction following exposure to a high-potency antipsychotic.

Outcome and follow-up

This child had no known lasting sequelae from catatonia. He returned to baseline functioning within 36 hours. He progressed in his treatment and was heading toward a slow transition to a less restrictive environment (home with great-grandmother with supports in place) when his guardian did not return him from a home visit, against medical advice. This author made multiple attempts (via telephone, letter and email) to contact the patient’s great-grandmother to discuss her reasons for withdrawing the child from treatment and to obtain consent for publication of this case report. All communication attempts were unsuccessful. This author also contacted the state’s DHR to discuss the case and was informed that the child’s great-grandmother moved him to a different state to live with extended relatives and that the child was missing.