Trauma-induced Catatonia in Pediatric Patients: Case Series and Literature Review

Abstract

Catatonia is a psychomotor syndrome that can present secondary to mood and psychotic disorders, other medical conditions, antipsychotic use, and alcohol withdrawal (Rasmussen et al., World Journal of Psychiatry, 6:391–398, 2016). In addition, acute and chronic trauma are increasingly recognized as a substrate for catatonia (Dhossche et al., Acta Psychiatrica Scandinavica, 125:25–32, 2012). Catatonia is a clinical diagnosis that relies on standardized examination of the patient. The Pediatric Catatonia Rating Scale (PCRS) was modified from the Bush Francis Catatonia Rating Scale and validated in children and adolescents (Benarous et al., Schizophrenia Research, 176:378–386, 2016). Changes included the addition of urinary incontinence, schizophasia and acrocyanosis; and withdrawal was separated into refusal to eat or drink and social withdrawal (Benarous et al., Schizophrenia Research, 176:378–386, 2016). In both pediatric and adult patients, catatonia must be differentiated from other movement disorder emergencies such as serotonin syndrome and neuroleptic malignant syndrome, which can also present with altered mental status and autonomic dysfunction (Rajan et al., Seminars in Neurology, 39:125–136, 2019). In pediatric patients, catatonia may be the genuine diagnosis in cases of Resignation Syndrome and Pervasive Refusal Syndrome (Sallin et al., Frontiers in Behavioral Neuroscience, 10(7), 2016; Ngo and Hodes, Clinical Child Psychology and Psychiatry, 25:227–241, 2019). The literature on pediatric catatonia is scarce but nevertheless expanding. Herein, we contribute to the literature by reporting four complex cases of pediatric catatonia that appear to have been triggered by a traumatic event and further reviewing the role of acute and chronic trauma in the presentation of pediatric catatonia.

Introduction

Catatonia is a psychomotor syndrome characterized by motor abnormalities and withdrawal (Burrow et al., 2023). It is commonly thought that catatonia can occur secondary to mood and psychotic disorders, other medical conditions, antipsychotic use, and alcohol withdrawal (Rasmussen et al., 2016). In addition, acute and chronic trauma are increasingly recognized as a substrate for catatonia (Dhossche et al., 2012; Dunn et al., 2017). The final common pathway is thought to involve dysfunction in neurotransmission of glutamate and gamma-aminobutyric acid (GABA) (Ariza-Salamanca et al., 2022; Benarous et al., 2018). Stuporous catatonia and excited catatonia due to any cause may progress to malignant catatonia (Rajan et al., 2019).

Catatonia is a movement disorder emergency and is likely underdiagnosed and undertreated in pediatric patients (Rajan et al., 2019; Luccarelli et al., 2022). This is urgently important because catatonia can have severe and even fatal outcomes. Medical complications of catatonia include dehydration, malnutrition, decubiti, contractures, aspiration pneumonia, and thromboembolic disease. Autonomic dysregulation in malignant catatonia can be lethal (Rasmussen et al., 2016). Early diagnosis and treatment of catatonia is therefore critical, especially in pediatric patients.

Catatonia is a clinical diagnosis that relies on standardized examination of the patient. The fifth edition of the Diagnostic and Statistical Manual of Mental Disorders defines catatonia as the presence of three or more of the following: stupor/immobility, catalepsy, waxy flexibility, mutism, negativism, posturing, mannerism, stereotypy, agitation, grimacing, echolalia, and echopraxia (American Psychiatric Association, 2013). The most frequent signs of catatonia are mutism, immobility, withdrawal, and staring (Rasmussen et al., 2016). In practice, the Bush-Francis Catatonia Rating Scale (BFCRS) has been the standard instrument for detecting catatonia and grading its severity since its publication in 1996 (Bush et al., 1996; Wortzel & Oldham, 2022). The Pediatric Catatonia Rating Scale (PCRS) was modified from the BFCRS and validated in children and adolescents two decades later (Benarous et al., 2016). The following are some of the changes that were made: urinary incontinence, schizophasia, and acrocyanosis were added; and withdrawal was separated into refusal to eat or drink and social withdrawal (Benarous et al., 2016; Ridgeway & Okoye, 2021). Urinary incontinence refers to involuntary leakage of urine; schizophasia refers to confused and unintelligible language; and acrocyanosis refers to bluish color in the hands and feet and around the mouth, reflecting vasomotor changes as seen in newborns and distressed infants. As this suggests, the signs and symptoms of pediatric catatonia are both physical and behavioral and often regressive.

In both pediatric and adult patients, catatonia must be differentiated from other movement disorder emergencies such as serotonin syndrome and neuroleptic malignant syndrome, which can also present with altered mental status and autonomic dysfunction. Serotonin syndrome, also referred to as serotonin toxicity, is associated with increased serotonergic activity in the central nervous system. It is seen with therapeutic medication use, inadvertent interactions between drugs, and intentional self-poisoning. Serotonin syndrome classically presents with the triad of mental status changes, autonomic hyperactivity, and neuromuscular abnormalities, most commonly hyperreflexia and clonus. Serotonin syndrome can therefore be differentiated from catatonia based on hyperreflexia and clonus and the recent use of a serotonergic agonist (Rajan et al., 2019).

Neuroleptic malignant syndrome is associated with decreased dopaminergic activity secondary to dopamine antagonists, namely antipsychotics (also called neuroleptics) and certain antiemetics. Neuroleptic malignant syndrome presents with the tetrad of mental status changes, muscular rigidity, fever, and autonomic instability. Neuroleptic malignant syndrome may represent a distinct pathological entity or a form of neuroleptic-induced catatonia (NIC). Catatonia, including NIC, can be differentiated from neuroleptic malignant syndrome based on a constellation of catatonic symptoms and response to lorazepam (Rajan, et al., 2019). In contrast, neuroleptic malignant syndrome may remit with antipsychotic discontinuation and supportive measures; moderate to severe cases are managed with dopamine agonists or dantrolene, which act downstream of lorazepam in reducing muscle tone (Stahl, 2013).

It is commonly recognized that an elevated creatine kinase (CK) level supports the diagnosis of catatonia (Fink & Taylor, 2003; Northoff et al., 1996). A more recent development in the field is the use of serum CK in the detection of emerging catatonia. CK may predict the emergence of catatonia, and CK trends positively correlate with progressive severity and resolution (Jaradeh et al., 2022). In using CK to support a diagnosis of catatonia, there are limitations in sensitivity, as there are cases of patients with catatonia without elevated CK. There are also limitations in specificity, as CK levels are also elevated in neuroleptic malignant syndrome and strenuous exercise, among other conditions. However, in the context of catatonic symptoms, an elevated CK level has a considerable positive predictive value and can confirm the diagnosis and differentiate catatonia from adverse antipsychotic effects such as acute dystonia (Jaradeh et al., 2022). As mentioned above, differentiation from neuroleptic malignant syndrome is based on catatonic symptoms and their response to a lorazepam challenge (Rajan et al., 2019; Ridgeway et al., 2021).

The core treatment of catatonia is medication management with a benzodiazepine, typically lorazepam; high doses are used for acute management and a tapering schedule is used for maintenance treatment (Rogers et al., 2023). Adjunctive management of catatonia can include antiepileptic agents, such as valproic acid and oxcarbazepine, but the efficacy of these is understudied. Ultimately, electroconvulsive therapy is indicated when increased doses of lorazepam do not result in improvement of the catatonic state, or when a patient requires immediate treatment due to concern for acute morbidity (Fink, 2009).

The efficacy of benzodiazepines such as lorazepam in the treatment of catatonia reflects the neurobiology of this psychomotor syndrome. Catatonia is thought to result from interconnectivity breakdown and imbalance in cortico-striato-thalamo-cortical circuits, rather than from a focal lesion in the central nervous system (Ariza-Salamanca et al., 2022). The main neurotransmitters in the pathophysiology of catatonia are glutamate, GABA, and dopamine. Stuporous catatonia in particular is thought to result from GABA-glutamate imbalance in critical circuits. Hypofunctioning of GABA_A receptors may result from intrinsic dysfunction or secondary to excess glutamate or NMDA receptor activity (Ariza-Salamanca et al., 2022).

It stands to reason that any factor that decreases GABA function would predispose to the development of catatonia or precipitate the syndrome. One such factor that has only recently started to receive attention is trauma (Dhossche et al., 2012). There are several connections between the neurobiology of catatonia and trauma. For example, developmental trauma is associated with hypothalamus–pituitary–adrenal (HPA) axis upregulation: baseline cortisol is elevated, and there is greater increase and slower decline of cortisol following stress exposure (Cross et al., 2017). GABA function has a central role in the integration of HPA stress responses in the basal forebrain and hypothalamus (Dhossche et al., 2012). Decreased GABA function is seen in developmental trauma and post-traumatic stress disorder, as in catatonia (Cross et al., 2017; Dhossche et al., 2012; Dunn et al., 2017; Huang et al., 2023). Another commonality between catatonia and trauma is increased neuroinflammation and oxidative stress (Dhossche et al., 2012). These associations suggest that developmental trauma may lower the threshold for the development of catatonia and acute trauma may induce catatonia. Moreover, these associations are consistent with our clinical understanding of catatonia and trauma, particularly in children and adolescent patients.

The Literature on Trauma-induced Pediatric Catatonia

Pediatric catatonia is understudied, but the scarce literature is nevertheless expanding. Indeed, in recent years, progress has been made in delineating catatonia in children and adolescents across a wide range of disorders. Pediatric catatonia has been associated with genetic, neurodevelopmental, mood, and psychotic disorders (Dhossche & Wachtel, 2010; Dhossche et al., 2010; Sorg et al., 2018). Moreover, there is increasing recognition that trauma may confer vulnerability to the development of catatonia, and may independently serve as a substrate for catatonia (Ahmed et al., 2021; Biles et al., 2021; Dhossche et al., 2012; Dunn et al., 2017; Giggie, 2021; Hortenstine & Youssef, 2020). In our formulation, deprivation and chronic trauma can predispose, and acute trauma can precipitate catatonia.

Understanding the factors that contribute to the presentation of catatonia is critical for better recognition and management. It is imperative to understand the role of trauma given the increase in cumulative and acute trauma in pediatric patients since the SARS-CoV-2 / Covid-19 pandemic (Fegert et al., 2020; Gindt et al., 2021), and the association of the pandemic with catatonia in vulnerable patients (Dell'Osso et al., 2021; Lynch & Bastiampillai, 2021). In this context, we evaluate four complex cases of trauma-induced pediatric catatonia treated at Loyola University Medical Center (LUMC) to delineate the role of trauma. These four patients presented within a two-month period in 2023.

The first case we report is a 5-year-old male with catatonia and extrapyramidal symptoms precipitated by excessive antipsychotic medication use in the setting of chronic trauma and attention-deficit/hyperactivity disorder. The second case is a 17-year-old transgender male with catatonia precipitated by antipsychotic medication use in the setting of ongoing bullying from peers and rejection from family; this is the second case in the literature of catatonia in a transgender adolescent. The third case is a 17-year-old male with catatonia and a first manic episode with psychotic features, arising in a background of sexual trauma and bullying. The fourth case is a 15-year-old female with chronic interpersonal trauma and severe acute sexual and emotional traumata who presented with catatonia. Taken together, the four cases we report demonstrate important challenges in diagnosing and managing pediatric catatonia, and reveal the complex role of trauma in the presentation of pediatric catatonia (Table 1).

Table 1.

Summary of cases of trauma-induced pediatric catatonia seen at LUMC. IM, intramuscular

Case	Patient Description	Predisposing factors	Precipitating factors (breaking point)	Perpetuating factors
1	5-year-old male	Chronic trauma exposure; neurodivergence (attention-deficit/hyperactivity disorder)	Haloperidol 5 mg IM once for acute agitation, quetiapine 25 mg twice daily, in the setting of psychiatric hospitalization	Limited due to early recognition, treatment, and collaboration with family
2	17-year-old transgender male	Chronic bullying from peers and shunning from family due to his transgender identity; sexual abuse in childhood; history of major depressive disorder and non-suicidal self-injury	Culmination of emotional turmoil from gender dysphoria and social rejection; haloperidol 15 mg IM within 15 min in the setting of emergent stabilization after a collision with a motor vehicle	Limited due to early recognition and treatment once seen in our emergency department
3	17-year-old male	Chronic bullying from peers; family member with bipolar disorder	Culmination of stress from bullying; sexual trauma	Premature discontinuation of lorazepam at the initial hospital; delayed diagnosis; haloperidol 5 mg IM once; scheduled risperidone 1 mg daily
4	15-year-old female	Chronic interpersonal trauma with mother; mother with schizoaffective disorder; chronic trauma exposure in her neighborhood; isolation from not attending school	Sexual and physical assault, verbal abuse from ex-boyfriend	Non-adherence to oral lorazepam maintenance treatment at an outside psychiatric hospital; interactions with mother

Case Series

Case 1

Case Description

Patient A is a 5-year-old male with recently diagnosed attention-deficit/hyperactivity disorder (ADHD), and with a history of chronic trauma exposure, who presented from an outside psychiatric hospital to the emergency department (ED) for evaluation of tachycardia, fatigue, and stiffness in the setting of having received multiple antipsychotic medications.

The patient had been admitted to the outside psychiatric hospital twelve days prior for aggressive outbursts and altercations with peers. He was started on a regimen intended to manage ADHD and aggression. He was specifically started on atomoxetine 20 mg daily, clonidine 0.05 mg twice daily, and quetiapine 25 mg twice daily. When he had an altercation with another child, he was given haloperidol 5 mg intramuscular (IM) injection. The patient was reported to be drowsy and with poor oral intake for the rest of the day. ​The next morning, the patient was noted to have tachycardia at 160 beats per minute, generalized rigidity, and facial masking. He was given diphenhydramine 25 mg IM to target extrapyramidal symptoms and was sent to our ED.

In our ED, the patient demonstrated intermittent tongue protrusion, fixed bilateral upward gaze, and facial and right upper extremity rigidity. ​His orofacial symptoms initially subsided but returned, requiring a total of diphenhydramine 48 mg intravenously (IV) per the recommendation of Poison Control. Although the patient remained tachycardic, he was afebrile. Laboratory results were significant for an elevated serum creatine kinase (CK) at 1,532 U/L.

Given the patient’s partial response to diphenhydramine and his persistent right upper extremity rigidity, a preliminary diagnosis was made of neuroleptic-induced acute dystonia and rule out catatonia. ​The patient received lorazepam intravenously (IV) with a 0.5 mg loading dose and 0.25 mg every six hours maintenance dose. Over the next twenty-four hours, diphenhydramine was tapered, his rigidity resolved, he became more alert, and his appetite was restored. A one-month lorazepam tapering schedule was initiated, and the patient was discharged home with resources on intensive outpatient mental health services.

Case Discussion

Catatonia must be differentiated from extrapyramidal symptoms induced by Dopamine type 2 (D2) receptor antagonism in the nigrostriatal pathway. It is important to recognize that extrapyramidal symptoms and catatonia may simultaneously occur and require different treatment. This was exemplified in this first case: the patient presented with concurrent acute dystonia and catatonia; following discontinuation of all antipsychotics, he partially responded to diphenhydramine treatment of acute dystonia and required lorazepam for treatment of catatonia. Given the patient’s partial response to diphenhydramine and full response with the addition of lorazepam, a final diagnosis was made of neuroleptic-induced acute dystonia and catatonia.​

Our initial impression focused on the patient’s muscle spasms and rigidity separately from his tachycardia. The patient’s motor symptoms largely resolved with diphenhydramine. Specifically, his tongue spasms and fixed upward gaze resolved, and this response is consistent with neuroleptic-induced acute dystonia. ​However, the patient’s persistent right upper extremity rigidity indicated an incomplete response to diphenhydramine. This incomplete response was in the setting of documented mixed antipsychotic medication use and an elevated serum creatine kinase (CK) level, which supports a clinical picture concerning for catatonia.​

The patient’s tachycardia could be an indicator of autonomic instability, which is seen in malignant catatonia and other acute movement disorders such as neuroleptic malignant syndrome. ​However, tachycardia could also be attributed to clonidine withdrawal. In this context, the lack of fever was reassuring against progression of catatonia to malignant catatonia, which may have occurred without early detection and treatment.

The present case is one of the youngest cases of pediatric catatonia in the literature (Leroy et al., 2018). The 5-year-old patient had early childhood exposure to trauma at home, witnessing interpersonal violence, mental illness, and substance abuse exacerbated by the Covid-19 pandemic. In our understanding, trauma early in life conferred vulnerability, and excessive antipsychotic use precipitated catatonia.

Case 2

Case Description

Patient B is a 17-year-old transgender male with a past psychiatric history of major depressive disorder and self-injurious behavior of cutting, who was brought by Emergency Medical Services (EMS) to our ED after a motor vehicle collision between an automobile and the patient on his bicycle. Per EMS, the patient was briefly cooperative in the ambulance but became hyperactive and combative in the ED, requiring a total of lorazepam 4 mg IM injections and haloperidol 15 mg IM injections over a course of 15 min. This initial combativeness raised the possibility of excited catatonia. The patient was ultimately intubated in the ED due to increased combativeness and admitted to the surgical intensive care unit for management. Work-up was negative for acute traumatic injuries except for a left subgaleal hematoma and scalp laceration seen on computerized tomography of the head (CTH). On the second day of hospitalization, the patient was extubated; vital signs remained stable, and the patient was alert and fully oriented.

On the third day of hospitalization, the rapid response team was called to the patient for the following motoric symptoms: involuntary tongue protrusion, jaw clenching, neck hyperextension, and head rotation to the right; fixed gaze with decreased blinking and minimal or no scanning of the environment; and pacing in the hospital room. Vital signs were stable, and the patient remained alert and fully oriented with intact sensorimotor examination. Neurology and Psychiatry services were consulted for evaluation.

CTH showed no acute changes, and the patient was given diphenhydramine 50 mg liquid suspension and started on an IV maintenance dose for suspected acute dystonia. However, there was scarce improvement in symptoms in the hour following diphenhydramine administration. Based on this, our Psychiatry team suspected neuroleptic-induced or worsened catatonia and administered a lorazepam challenge with lorazepam 1 mg IV. There was a positive response to lorazepam, with resolution of motoric symptoms that had masqueraded as acute dystonia.

The patient was therefore started on lorazepam 1 mg IV every six hours. Stat and daily serum CK were recommended to support the diagnosis of catatonia and trend treatment response. Stat CK was not obtained, but the following morning CK resulted at 854 U/L. CK decreased with treatment, resulting at 484 U/L after 48 h. The patient continued to improve without recurrence of the aforementioned catatonic symptoms. He was ultimately discharged home with a gradual lorazepam taper.

Of note, on initial psychiatric interview, the patient endorsed depressive symptoms and reported abstinence from non-suicidal self-injurious behavior for nearly one month. The patient disclosed but did not elaborate on sexual abuse he experienced as a child by a neighbor. He also reported emotional turmoil related to gender dysphoria and disapproval from family and peers. For these reasons, before discharge, the patient was strongly encouraged to establish care with mental health services.

Case Discussion

This case is similar to the first in our case series in that catatonia needed to be differentiated from acute dystonia. However, this case was consistent with catatonia and not concurrent acute dystonia. The rapid response was called two days after the patient last received haloperidol, whereas acute dystonia commonly presents within hours of the most recent antipsychotic dose. In addition, there was no response to diphenhydramine in the hour prior to the initiation of lorazepam treatment, and catatonic symptoms remitted with lorazepam. Moreover, in the setting of catatonic symptoms, serum CK was elevated: this is consistent with central GABAergic hypofunction and generalized increased muscle tone, rather than discrete muscular spasm. The patient was therefore diagnosed with catatonia.

There was a vulnerability underlying the presentation seen in our emergency department. It seemed to our treatment team that the patient was predisposed to catatonia by unspecified childhood abuse and recent cumulative trauma. Specifically, the patient experienced bullying from peers and shunning from family as a result of his transgender identity. This is the second case in the literature of catatonia in transgender youth (Maffei & Dean, 2021).

Case 3

Case Description

Patient C is a 17-year-old male with no known psychiatric history, no known substance use, a past medical history of asthma, and new-onset unspecified psychosis. One month prior to presenting to our hospital, the patient presented to an outside ED with mutism, decreased oral intake, staring, declining school performance, and marked social withdrawal for one year. The patient reported bullying at school. The patient demonstrated improvement with a lorazepam challenge and was transferred to a nearby hospital for further medical work-up and management. At the hospital, another lorazepam challenge was considered but deemed unnecessary as the patient became more interactive, and he was discharged without medications.

About 20 days later, the patient again demonstrated marked withdrawal and was subsequently admitted to a psychiatric hospital where he was started on risperidone. However, due to elevated CK in the setting of tachycardia and hypertension, the patient was transferred to our ED. The patient was afebrile but had a serum CK level of 5,653 U/L, heart rate of 108 beats per minute (bpm), and a blood pressure of 155/74 mm of mercury (mmHg). The patient was notably agitated and hyperactive, with fearful mood, tangential thought process, inattentiveness, and impulsivity. As a result, the patient was given haloperidol 5 mg IM injection and midazolam 2 mg IM injection. Complete metabolic panel and urinalysis were reassuring against the development of acute kidney injury secondary to rhabdomyolysis. For management of psychosis, the patient was given risperidone 1 mg twice daily and lorazepam 1 mg every six hours as needed for agitation. That evening, the patient was reported to be sexually acting out, exposing his genitals to staff and requiring restraints.

On psychiatric evaluation, the patient continued to demonstrate sexual preoccupation; however, his appetite and social withdrawal were improving. Given the concern for catatonia with markedly elevated CK, and given the patient’s improvement with lorazepam, the patient was started on a regimen of lorazepam 1 mg IV every six hours. Risperidone was discontinued. Early in the acute management of catatonia, the patient endorsed a recent history of significant bullying at school and made vague statements about sexual trauma that he experienced at an unclear point in the past.

As catatonic symptoms resolved, manic and psychotic symptoms became more prominent: the patient had an expansive and labile mood, grandiosity, rapid speech, psychomotor agitation with arrhythmic leg shaking, and paranoid delusions. To target his mood and impulsivity, valproic acid was started and titrated up to 500 mg twice daily. Later, due to hyperammonemia at 126 ug/dL, valproic acid was discontinued despite improvement in symptoms consistent with an acute manic episode with psychotic features. Lorazepam was titrated up to 2 mg IV every six hours. The differential diagnosis at this time included bipolar spectrum versus schizophrenia spectrum disorder in addition to catatonia. It was notable that the patient’s manic symptoms were in the context of significant benzodiazepine doses required in the treatment of catatonia.

The patient’s CK continued to downtrend and the patient’s scheduled lorazepam 2 mg IV every six hours was changed to oral formulation with a plan to taper as an outpatient. To manage the patient’s mood and behavioral symptoms without the risk of inducing hyperammonemia and without dopamine blockade, oxcarbazepine 300 mg twice daily was started. The patient was discharged to a psychiatric hospital with a lorazepam taper, with the recommendation to avoid antipsychotic agents with potential consideration of partial dopaminergic agonists. The leading diagnoses at discharge were resolved catatonia and bipolar disorder—type I, with a first episode of mania with psychosis.

Case Discussion

The patient in this case had a one-year prodrome of mutism, poor oral intake, social withdrawal, and a steep decline in functioning. While such a period is typically associated with schizophrenia spectrum disorders, it may represent an emerging catatonic process that can worsen with antipsychotic medication use, as in our patient. In fact, in this case, catatonia had likely been perpetuated not only by initiation of risperidone, but also by prior discontinuation of lorazepam. In our emergency department, the patient was suspected to have catatonia based on his history and elevated CK, and catatonia was confirmed with a lorazepam challenge.

In pediatric patients, catatonic symptoms may precede more classic psychiatric symptoms (Sorg et al., 2018). In this case, catatonia preceded the emergence of mania; mood and psychotic symptoms only fully emerged with treatment of catatonia. Additionally, the patient’s response to oxcarbazepine adds to the literature on oxcarbazepine in the management of catatonia, and oxcarbazepine discontinuation as a precipitant of catatonia. Catatonia was treated not only with lorazepam, but also with oxcarbazepine augmentation; this adds to the literature on oxcarbazepine in the management of catatonia, and oxcarbazepine discontinuation as a precipitant of catatonia (Smith et al., 2023; Zwiebel et al., 2019).

The patient in this case was relatively young for the onset of bipolar disorder—type I. In our understanding, the patient’s trauma history and ongoing psychosocial stressors accelerated the development of his illness and put him at risk for catatonia. In this light, the patient’s sexually inappropriate comments and gestures can be understood as a reaction to unspecified sexual trauma and chronic bullying. Moreover, the prominence of sexual preoccupation in this case is interesting given that affective disturbances and behavioral problems such as nudism are considered part of the catatonic syndrome by some authors who rely on Kahlbaum’s initial conceptualizations (Ahmed et al., 2021; Fink, 2013; Northoff et al., 1999).

Case 4

Case Description

Patient D is a 15-year-old female with an unremarkable past medical history who presented from an outside psychiatric hospital due to concerns for catatonia. Prior to psychiatric hospitalization, the patient engaged in bizarre behavior for two months, including episodes of the patient pacing around the room, standing, and staring. Additionally, the patient was reported to have paranoid thoughts, visual and auditory hallucinations, and suicidal ideation. The patient had a history of chronic interpersonal trauma from interactions with her mother, as well as severe sexual and physical trauma from her ex-boyfriend which closely preceded this episode, and which required treatment for sexually transmitted infections.

The patient had been at the psychiatric hospital for three days and during that time she had no oral intake and became more verbally and physically withdrawn. By the time the patient presented to our ED, she was mute, staring, and urinating and defecating on herself. Laboratory results were significant for elevated CK of 644 U/L. Given the concern for catatonia, the patient was started on lorazepam and the patient’s symptoms of akinetic catatonia were resolved, including CK levels. She was discharged back to the inpatient psychiatric hospital after lorazepam was tapered and changed to oral formulation, with a plan for further tapering while an inpatient. However, after a few days the patient returned to our hospital due to non-adherence to lorazepam maintenance treatment, resulting in no oral intake and increased psychomotor slowing. The patient additionally had somatic concerns such as abdominal pain and leg pain, for which diagnostic work-up found no organic etiology.

The patient was restarted on lorazepam 2 mg IV every six hours with moderate improvement in oral intake but with signs of staring and mutism. An attempt to switch to oral formulation was unsuccessful due to inconsistent adherence, and even with intravenous treatment, clinical improvement plateaued at a state of severe psychomotor retardation, social withdrawal, mutism with occasional schizophasia, and delusions. Given the patient’s catatonic state, medication non-adherence with concern for conversion to resistance, and severe trauma history, we recommended transfer to an outside hospital for electroconvulsive therapy (ECT). The patient’s mother declined ECT treatment for the patient. The patient continued to receive treatment with high doses of lorazepam with gradual improvement in catatonic symptoms punctuated by episodic acute decompensation when her mother failed to visit or visited while intoxicated with alcohol. Her mother did not predictably or consistently visit the patient and demonstrated high expressed emotion when present.

As the patient recovered from a critical catatonic state, more of her trauma history came to light. For example, the treatment team learned that the patient resides with her maternal grandmother and two older brothers in a Chicago neighborhood with marked poverty and violence, with her mother intermittently present. The patient had been unable to attend school the prior year due to a lack of transportation. About two months prior to presenting to our hospital, the patient and her brothers lost two of their close friends to gun violence. At the time, the patient was in an abusive relationship wherein she was sexually and physically assaulted and verbally abused. This traumatic time corresponds to the onset of the patient’s catatonic behavior, as reported by her grandmother.

In consideration of the above psychosocial history, the Department of Child and Family Services escalated their priority of this case. As their investigation continued, legal guardianship of the patient was transferred from her mother to her grandmother. During this time, the patient tolerated a switch from intravenous to oral lorazepam. The treatment team attempted to find placement for the patient in a psychiatric ward without success. However, the team noticed a marked improvement in the patient after her grandmother became her guardian. The patient was ultimately discharged home with her grandmother with referral to an intensive outpatient program to continue management of her catatonia and trauma reaction.

Case Discussion

Although catatonia may present similarly in pediatric patients compared to adults, there are notable differences seen in this case and reflected in the adaptation of the BFCRS to pediatric patients (Benarous et al., 2016; Ridgeway & Okoye, 2021). Specifically, we noted that the patient had added symptoms of urinary incontinence and schizophasia, and that acrocyanosis may present later, with more severe arterial desaturation, in patients with dark skin. Furthermore, the patient had prominent social withdrawal and reduced oral intake; she became underweight and nearly required nasogastric intubation and feeding on more than one occasion. The patient thus had failure to thrive and psychogenic catatonia, induced by complex and acute trauma.

It is moreover notable that in addition to catatonic symptoms, the patient had psychosomatic symptoms. In the formulation of the treatment team, the patient experienced chronic interpersonal trauma and lived under the threat of violence in her neighborhood, which conferred vulnerability to catatonia in the form of baseline stress reactivity and neuroimmunomodulation. The patient then experienced severe sexual and physical abuse, and her acute trauma reaction presented as catatonia and somatization.

Discussion

Trauma played a role in the presentation of catatonia in each case in this series with subtle but important differences (Table 1). These differences support the view that adverse childhood experiences and trauma can predispose to catatonia, and acute trauma can precipitate catatonia, within children and adolescents (Fig. 1).

Fig. 1.

Predisposing, precipitating, and perpetuating factors in the presentation of catatonia. Aps, antipsychotic medications

Trauma Can Confer Vulnerability to Catatonia

Several lines of evidence suggest that trauma exposure can confer vulnerability to catatonia. For example, Ross and Browning (2016) studied 100 inpatients in a hospital trauma program with the BFCRS and the Adverse Childhood Experiences (ACE) questionnaire. In this sample, catatonic symptoms were frequent, and there was a significant positive correlation between BFCRS scores and ACE questionnaire scores. Consistent with this, the 5-year-old male presented first in this series had early childhood exposure to trauma that predisposed him to the development of catatonia; given this vulnerability, excessive antipsychotic medication use precipitated catatonia rather than an uncomplicated acute dystonic reaction. A similar case report described a 10-year-old male in psychiatric residential treatment with early childhood neglect and perinatal exposures that predisposed him to neuroleptic-induced catatonia (NIC). In this patient, catatonic symptoms emerged 24 h after receiving a first-time dose of haloperidol and improved with lorazepam (Giggie, 2021). These cases suggest that childhood maltreatment in its variant forms may be a risk factor for the development of catatonia in children.

Cumulative trauma may also confer vulnerability to catatonia in adolescence. For example, in the second case in this series, a 17-year-old transgender male developed catatonia in the setting of chronic bullying from peers and shunning from family as a result of his transgender identity. Similarly, cumulative trauma from chronic bullying was thought to contribute to the presentation of catatonia in Case 3, the 17-year-old male with catatonia and first-time acute mania with psychotic features. This compares interestingly to a case report of post-traumatic catatonia, which described a 14-year-old female with no prior psychiatric history who was admitted for disorganized behavior and decreased need for sleep in the setting of significant cyberbullying from her schoolmate, and who developed frank catatonia in response to risperidone (Dhossche et al., 2013). These cases suggest that experiencing bullying and rejection can contribute to a predisposition to catatonia.

In these cases, organic etiologies for catatonia were excluded, and catatonia was therefore considered a primary psychiatric disorder. However, there are cases in the literature of trauma conferring vulnerability to catatonia secondary to an organic etiology, specifically anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis. For example, a recent case report described a 14-year-old female with catatonia secondary to anti-NMDAR encephalitis that developed within one week of a sexual assault (Bogden et al., 2022). The authors considered the possibility that recent trauma conferred vulnerability to autoimmune encephalitis with catatonic symptoms, as stress can cause immune dysregulation (Bogden et al., 2022; Obi et al., 2019). Another case report described a 12-year-old transgender male that presented with altered mental status, agitation, and suicidal ideation in the setting of recent disclosure of his gender identity to family and classmates without their acceptance (Maffei & Dean, 2021). Unfortunately, organic etiologies for altered mental status were not initially considered due to evident emotional stressors. Catatonic symptoms worsened in response to antipsychotic medications (chlorpromazine, haloperidol, and risperidone) before the patient was diagnosed with catatonia secondary to anti-NMDAR encephalitis (Maffei & Dean, 2021). These cases raise the possibility that traumatic experiences can contribute to catatonia that is organic in nature, underscoring the importance of a comprehensive biopsychosocial approach.

The role of complex trauma in the presentation of catatonia in adolescence is best examined in Case 4, the 15-year-old patient with a protracted course of catatonia and somatic symptoms. The patient was suspected to have complex post-traumatic stress disorder given intermittent neglect and high expressed emotion from her mother, and given the pattern of regression in oral intake and withdrawal following interactions with her mother. It seemed that chronic interpersonal trauma predisposed the patient to the development of catatonia and impeded recovery. Relevant to this discussion, Micaldi et al. (2021) reported a case of a 16-year-old female in state custody with catatonia and schizoaffective disorder, and with a history of unspecified trauma starting at an early age. Complex post-traumatic stress disorder was considered for this patient, and she had catatonia that worsened when she resumed contact with her father. This case serves as further support for the role of complex trauma in conferring vulnerability to catatonia and influencing its course.

Acute Trauma Can Precipitate Catatonia

Acute traumatic events can precipitate catatonia in susceptible patients. This is seen in cases with unspecified trauma. For example, the 17-year-old male in Case 3 had a history of unspecified sexual trauma that may have precipitated his development of catatonia and bipolar disorder with psychotic features, and that may have contributed to his sexual preoccupation.

There are scarce case reports of specified traumatic events that closely precede the onset of catatonia, but these reports further support the view that acute trauma can precipitate catatonia in susceptible patients. For example, the 15-year-old female in Case 4 experienced sexual and physical abuse from her ex-boyfriend that closely preceded her development of catatonia and somatic symptoms. Another case report described a 12-year-old female with catatonia that was precipitated by two life-threatening events within two weeks, and that severely worsened before the patient was brought to psychiatric care. The patient was diagnosed with post-traumatic stress disorder associated with catatonia (Ahmed et al., 2021).

Dunn et al. (2017) reported a case of catatonia and psychotic symptoms secondary to the trauma of captivity in a cult environment in late adolescence. The patient was 16 years old when she was lured and coerced by a cult leader; she was then held against her will for over four years. During this time, she experienced severe abuse, including being impregnated twice and losing her second child to profound deprivation and abuse. The patient was rescued and her functioning declined over the subsequent six months, at which time she was admitted for medical-psychiatric care. The patient was diagnosed with post-traumatic stress disorder associated with catatonia. In this case, catatonia resulted from the major trauma and abuse that the patient experienced when held captive (Dunn et al., 2017).

In a similar case reported nearly two decades prior, catatonia was seen as a recurrent extreme manifestation of post-traumatic stress disorder in a patient who had survived the Holocaust in late adolescence (Shiloh et al., 1995). In these cases, catatonia reflected the extreme severity of the avoidance, numbness, and motor responses encountered in post-traumatic stress disorder (Dunn et al., 2017; Shiloh et al., 1995). From this discussion, it is evident that reactions to acute trauma and traumatic recollections can present as catatonia.

Framework for the Role of Trauma in Catatonia

The observation that trauma can predispose to and precipitate pediatric catatonia is consistent with connections between the neurobiology of catatonia and trauma, as mentioned above (Cross et al., 2017; Huang et al., 2023; Dhossche et al., 2012; Dunn et al., 2017). In brief, chronic trauma in critical periods results in reduced GABAergic function and increased stress reactivity and neuroinflammation, which predisposes to catatonia (Cross et al., 2017). Acute trauma can precipitate catatonia, as catatonia results from GABA-glutamate imbalance within a pro-inflammatory environment (Ariza-Salamanca et al., 2022).

Evolutionary theories are also consistent with the view that trauma has a role in the presentation of catatonia. For example, catatonia can be conceptualized as a fear response in the face of inevitable and inescapable danger, akin to the animal defense strategy of tonic immobility. In this perspective, catatonia represents a common end state to impending doom or extreme stress (Moskowitz, 2004). This is congruent with an original conceptualization of catatonia in 1874, which gave trauma a central role in catatonia in many young adult cases (Biles et al., 2021; Dhossche et al., 2012; Dunn et al., 2017). Recent work on the phenomenological experience of catatonia centers on an atavistic fear of separation and the evolutionary need for secure attachments. In this perspective, early life trauma lowers the threshold for catatonic responses to severe stressors (Fricchione, 2023).

Furthermore, the association of trauma, neglect, and other adverse childhood experiences with pediatric catatonia is consistent with the theory that catatonia is the genuine diagnosis in Resignation Syndrome and Pervasive Refusal Syndrome, as both are associated with psychologically traumatized children and adolescents seeking asylum (Sallin et al., 2016; von Knorring & Hultcrantz, 2020; Ngo & Hodes, 2019). Cases of Pervasive Refusal Syndrome (PRS) present with passive or active refusal of oral intake, mobilization, speech, and attention to personal care; incontinence is also common (Ngo & Hodes, 2019). PRS presents similarly in asylum-seeking and non-asylum-seeking children, with cases sharing in common a history of traumatization. In fact, PRS was initially thought to be a form of post-traumatic stress disorder (Ngo & Hodes, 2019). A more recent reconceptualization, Pervasive Arousal Withdrawal Syndrome, posits that there is an underlying breakdown in the normal relationship between the parasympathetic and sympathetic nervous system (Ngo and Hodes, 2019). The clinical presentation and theorized neurobiology of PRS therefore is consistent with that of trauma-induced catatonia in children and adolescents.

Resignation Syndrome (RS) has been observed among refugee children in Sweden. It is characterized by a gradual withdrawal that progresses to deep stupor, immobility, mutism, incontinence, and inability to eat or drink, which requires tube feeding and assistance with activities of daily living (Sallin et al., 2016). RS therefore presents similarly to stuporous catatonia in children and adolescents (Benarous et al., 2016). There are especially notable parallels between cases of RS and Case 4. The patient in Case 4 had a history of cumulative trauma exposure and acute abuse; she presented with withdrawal, severe psychomotor retardation, mutism, incontinence, and refusal of oral intake, and nearly required nasogastric tube feeding. Given that RS presents similarly to catatonia but has a circumscribed geographic distribution, it has been proposed that RS represents a culturally-bound form of psychogenic catatonia (Sallin et al., 2016).

Kozlowska et al. (2021) similarly view “the shutdown state” seen in asylum-seeking children as an evolutionary and neurobiological response to trauma. According to the authors, the pattern of trauma experienced by asylum-seeking children involves cumulative adverse childhood events that have occurred at multiple time points across the family narrative and throughout development (Kozlowska et al., 2021). This is interesting in light of the cases presented above, wherein cumulative adverse events were common. It seems that psychogenic catatonia and the shutdown state seen in asylum-seeking children are not distinct clinical entities but rather a response to trauma that occurs within a personal, familial, and cultural context (Sallin et al., 2016; Ngo & Hodes, 2019; Kozlowska et al., 2021).

Naturally, trauma is one factor in the presentation of catatonia and must be understood within the context of other factors, which may include psychiatric, neurologic, and other medical conditions; withdrawal from anti-catatonic agents; use of antipsychotic agents; and barriers to treatment for catatonia (Fig. 1). These other factors interacted with trauma histories in the case series presented (Table 1) and in cases reported in the literature (Table 2). Given that trauma can lower the threshold for the development of catatonia, and given that trauma can independently serve as a substrate for catatonia, children and adolescents presenting with catatonia should be screened for trauma as part of a comprehensive medical work-up (Dhossche et al., 2012).

Table 2.

Summary of cases of trauma-induced pediatric catatonia reported in the literature and referenced in this review. IM, intramuscular

Case	Patient Description	Predisposing factors	Precipitating factors (breaking point)	Perpetuating factors
Ahmed et al., 2021	12-year-old female	None reported	Two life-threatening events within two weeks	Delayed presentation to medical treatment
Bogden et al., 2022	14-year-old female	Possible role of sexual assault that occurred five days prior to presentation, as stress can cause immune dysregulation	Anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis	Delayed diagnosis due to the need for sendout laboratory tests
Dhossche et al., 2013	14-year-old female	None reported	Significant cyberbullying from her schoolmate; risperidone 1 mg daily	None reported
Dunn et al., 2017	21-year-old female	Captivity in a cult environment from age 16–21; severe deprivation and abuse, including sexual assault, being impregnated, and giving birth to two children	Witnessing the death of her second child in infancy from severe deprivation and abuse	Limited due to early recognition, treatment, collaboration with family, and post-traumatic growth
Giggie, 2021	10-year-old male	State custody for abuse and neglect; intrauterine exposure to alcohol and multiple drugs; early childhood deprivation; oppositional defiant disorder	First-time haloperidol 5 mg IM for acute agitation in the setting of long-term psychiatric residential treatment	None reported
Maffei & Dean, 2021	12-year-old transgender male	None reported; possible role of bullying and shunning from peers and family, as stress can cause immune dysregulation	Anti-NMDAR encephalitis	Delayed consideration of organic etiologies in the setting of known psychosocial stressors; chlorpromazine, haloperidol, and risperidone
Micaldi et al., 2021	16-year-old female	State custody; sexual abuse from father; sexual and physical abuse in foster family; early maternal death to cancer	None reported	Scheduled risperidone; haloperidol IM once for acute agitation; resuming contact with father

Conclusion

In summary, our four cases support the conceptualization that trauma can predispose to and precipitate pediatric catatonia, and moreover that children and adolescents presenting with catatonia should be screened for trauma (Dhossche et al., 2012). The experience of adverse childhood events including trauma and neglect is pervasive in our society. Furthermore, transgender youth such as our second patient may be especially vulnerable given the current political climate and wave of anti-transgender legislation (Barbee et al., 2022). As seen in our fourth case and in the literature more broadly, children and adolescents with state involvement and those seeking asylum may also be especially vulnerable (Giggie, 2021; Micaldi et al., 2021; Sallin et al., 2016; von Knorring & Hultcrantz, 2020; Ngo & Hodes, 2019; Kozlowska et al., 2021). Given the increase in trauma and exacerbated psychiatric public health crisis since the Covid-19 pandemic, this case series urges us to raise our suspicion for pediatric catatonia and to expect an increase in its incidence, so as to improve patient care outcomes via timely identification and management (Fegert et al., 2020; Gindt et al., 2021).

Author Contributions

Data collection, literature review, and manuscript drafting and editing were completed by DRE, NT, TKP, and EM. All authors contributed to preliminary work, and all authors approved the final manuscript for submission.

Funding

This research project was not funded but rather undertaken out of care and interest in the field of trauma.

Data Availability

No supplemental or supporting data is available.

Declarations

Ethical Approval

This research project was reviewed and assigned a status of Exempt by the Institutional Review Board for the Protection of Human Subjects, Loyola University Health Sciences Division.

Competing Interests

The authors have no financial or non-financial competing interests.