CASE REPORT

published: 24 March 2021

doi: 10.3389/fpsyt.2021.637886

Case Report: A Case of Pediatric

Catatonia: Role of the Lorazepam
Challenge Test
Laura Ridgeway 1*, Albert Okoye 1*, Ian McClelland 2 , Dirk Dhossche 3 , Deniz Kutay 1 and
Mario Loureiro 1
1
Department of Child and Adolescent Psychiatry, University Hospital Waterford, Waterford, Ireland, 2 Department of Liaison
Psychiatry, Children’s University Hospital, Dublin, Ireland, 3 Department of Psychiatry and Human Behaviour, University of
Mississippi Medical Center, Jackson, MS, United States

A case of a 12-year-old boy who developed catatonia is presented. He had no previous

psychiatric history but has a family history of affective disorder. An extensive medical
Edited by: workup was negative. Despite a negative lorazepam challenge test, lorazepam was
David Cohen, titrated up to 24 mg/day, with resolution of most catatonic symptoms. The case highlights
Université Pierre et Marie
an important point in the management of catatonia that may be a source of confusion,
Curie, France
i.e., a positive lorazepam challenge test corroborates the diagnosis of catatonia; however,
Reviewed by:
Gábor Gazdag, a negative lorazepam challenge test does not negate the diagnosis of catatonia, and
Jahn Ferenc Dél-Pesti Kórház és subsequent focused benzodiazepine treatment may still be effective.
Rendelõintézet, Hungary
Xavier Benarous, Keywords: catatonia, paediatric, depression, lorazepam, catatonia behavior, child
Centre Hospitalier Universitaire de
Amiens, France
Diana Punko,
INTRODUCTION
Massachusetts General Hospital and
Harvard Medical School,
Catatonia is a potentially life-threatening but treatable condition that could be underdiagnosed and
United States
undertreated in children and adolescents (1–3). The prevalence of pediatric catatonia in psychiatric
*Correspondence:
clinics varies from 0.6 to 17% (4, 5). The majority of pediatric catatonic cases occur at pubertal ages
Laura Ridgeway
(6). In contrast to adults, catatonia in children and adolescents is more common in boys than girls
[email protected]
Albert Okoye with a ratio of 2:1 (7–9).
[email protected] For all diagnostic categories of catatonia, the Diagnostic and Statistical Manual of Mental
Disorders, Fifth Edition (DSM-5), requires the presence of 3 out of 12 symptoms (10). The
Specialty section: presenting symptoms of catatonia are thought to be similar across age ranges. Some symptoms that
This article was submitted to may be significant in younger patients include refusal to eat or drink, social withdrawal, repetitive
Child and Adolescent Psychiatry, movements, and regressive symptoms, such as urinary incontinence (4).
a section of the journal Clinical rating scales originally designed for adults with catatonia have been used in children
Frontiers in Psychiatry
to assist diagnosis and estimate severity, including the Bush–Francis Catatonia Rating Scale and
Received: 04 December 2020 the Modified Rogers Scale. Cohen also developed a modified Pediatric Catatonia Rating Scale from
Accepted: 03 February 2021 the Bush–Francis Catatonia Rating Scale (4, 11). A lorazepam challenge test can be considered for
Published: 24 March 2021
diagnostic validation of catatonia (5).
Citation:
Ridgeway L, Okoye A, McClelland I,
Dhossche D, Kutay D and Loureiro M
CASE REPORT
(2021) Case Report: A Case of
Pediatric Catatonia: Role of the A previously healthy 12-year-old boy (Boy A), presented to the pediatric emergency department
Lorazepam Challenge Test. with a 4-month history of progressive functional decline and social withdrawal, described by
Front. Psychiatry 12:637886. the parents as seeming “lost” and “distant.” He had a brief 2-week period of improvement in
doi: 10.3389/fpsyt.2021.637886 his symptoms 2 months previously during holidays from school. His symptoms also seemed to

Frontiers in Psychiatry | www.frontiersin.org 1 March 2021 | Volume 12 | Article 637886

Ridgeway et al. Case Report: Case of Pediatric Catatonia

worsen during weekdays and improve over the weekends when deemed an option at that stage as accessing ECT for a pediatric
he reportedly engaged in sports activity. The authors wondered patient in the Republic of Ireland carries many difficulties,
if this was due to worsening anxiety, low self-confidence, and including cultural barriers, the necessity of High Court approval,
poor concentration and attention in the increasingly cognitively the lack of an experienced center in providing ECT to pediatric
demanding academic setting. His speech had become minimal patients, and, at the time, a referral to a UK center was
with regressed behavior over the preceding 2 weeks, and complicated by the coronavirus (COVID-19) pandemic. Due to
he required assistance with dressing, washing, feeding, and continued refusal to eat, drink, or swallow prescribed fluoxetine
prompting to void urine. after several days of intravenous lorazepam 1 mg TDS, he was
Boy A was pre-morbidly “a very likable young boy” who started on IV fluids to prevent dehydration.
enjoyed sports with many friends in school and although could In addition to the investigations performed on his previous
be quiet with a “tendency to be anxious,” he had no prior mental admission, venous blood gas, ammonia, lactate, glucose,
health difficulties. He lived with his mother, father, and two creatinine kinase, and cerebrospinal fluid for N-methyl-D-
sisters. A recent significant stressor for the family was his father’s aspartate (NMDA) receptor antibodies were all within normal
diagnosis and two admissions in the previous 4 months for ranges. MRI scan of the brain and electroencephalogram were
bipolar affective disorder following a manic episode. At the time also unremarkable. The dietitian started nasogastric feeding
of presentation, his father’s mental health was stable. after 1 week of no oral intake and 3-kg weight loss, despite
Apart from a slow gait, the pediatric team’s physical and then being on lorazepam 10 mg daily in four divided doses.
neurological examination was within normal limits. He was Fluoxetine was then given via the nasogastric tube with daily
admitted to the pediatric medical ward on suspicion of an blood electrolyte monitoring due to risk of refeeding syndrome.
encephalopathy where he had a comprehensive medical workup, Clinically, however, his facial muscles appeared more relaxed and
including full blood count, liver function tests, C-reactive slightly emotionally expressive and he seemed to move his head,
protein, thyroid function tests, electrolytes, antistreptolysin-O- arms, and neck somewhat more easily. During physiotherapy
titer, lactate dehydrogenase, uric acid, CT scan of the brain, and sessions over the following few days, he could subsequently
urine toxicology. All studies were unremarkable. stand with assistance, move his arms, and raise both legs at his
Child and adolescent psychiatry consult was then sought, parent’s request. Waxy flexibility became gradually less evident,
and on mental state examination, Boy A appeared preoccupied, and he displayed non-verbal interaction with his parents, smiling
anxious, and irritable, with fleeting eye contact. His speech was hesitantly, and engaging in eye contact. Lorazepam had gradually
minimal, and he responded to some questions by shrugging been titrated up to 16 mg daily in four divided doses with no
his shoulders, but his engagement improved as the interview sedation or respiratory compromise by the end of the second
progressed. He had apparent difficulty and reluctance to move week, at which point Boy A could stand up and mobilize
his limbs, with some evidence of psychomotor slowing. While not independently during physiotherapy sessions. His parents also
initially reporting consistent symptoms of depression, his mood reported him saying a few words “mum,” “dad,” and he appeared
had been pervasively low for more than 2 weeks but no evidence able to swallow a few spoons of food, with noticeably reduced
of psychotic symptoms. The initial impression was depression salivation. He had gained 1.5 kg, which increased to 2.7 kg by
and anxiety with some evidence of psychomotor slowing. Boy A the end of the third week with improved dietary intake, and
was started on fluoxetine 10 mg OD for 1 week, to be increased nasogastric tube feeds then discontinued.
to 20 mg OD thereafter, and lorazepam 0.5 mg daily was added to Although there was still some evidence of psychomotor
relieve his anxiety and irritability. He was discharged home a few slowing, rigidity was less evident and his activity was more
days later, given the stability of his mental state and daily phone fluid such that he could now throw, kick, and catch a ball
reviews, and 1-week psychiatry clinic appointment was agreed. with his parents and was smiling and speaking more frequently.
On review in the clinic, his parents reported reduced oral He was however indecisive and appeared “stuck” and required
intake to small sips of fluids, salivating, and inability to swallow prompting when activity was interrupted. Persistence of these
oral medication. On urgent readmission to the pediatric medical observations informed further increase in the dose of lorazepam
ward, he showed little reactivity to the environment (stupor) to 18 mg by the fourth week of admission, which led to
and was essentially mute. There was evidence of psychomotor further improvement in psychomotor functioning and physical
retardation, catalepsy, waxy, flexibility, rigidity, some degree of activity, with still no evidence of sedation. With improvement in
posturing, and negativism. He also displayed stereotypies, such psychomotor functioning, however, Boy A became increasingly
as twitching and running his hands over things, mannerisms, more tearful and intensely emotional, expressing guilt for
and grimacing. His Bush–Francis Catatonia Rating Scale severity relatively minor misdemeanors and repeatedly apologizing to the
score was 30, and screening score was 11, but there was point of perseveration. He nevertheless was discharged home
no evidence of psychotic symptoms. His systemic vital signs at the end of the fourth week on lorazepam 18 mg daily and
were within normal limits. His diagnosis was conceptualized as fluoxetine 20 mg daily with a Bush–Francis Catatonia Rating
retarded catatonia likely secondary to a depressive illness. Scale severity score of 3 and screening score of 0. The follow-
A lorazepam challenge test [2 mg intravenously (IV)] showed up plan was for regular phone consultations, weekly psychiatric
no evidence of response. However, the decision was made clinic reviews, and psychology and occupational therapy input.
to continue titrating up the dose of lorazepam while closely His emotionality persisted with incessant unprovoked
monitoring response. Electroconvulsive therapy (ECT) was not tearfulness and weeping and “a great fear of everything,”

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Ridgeway et al. Case Report: Case of Pediatric Catatonia

including worries that bad things would happen. He was clingy pediatric catatonia (4, 5). Often, in cases with developmental
and remained apologetic, repeatedly thanking parents, “thank disorders, the diagnosis can be more difficult due to the overlap
you so much, you are best mother/father in the world.” He was of symptoms.
indecisive and frequently seeking reassurance and permission As shown in this case, there is not always a clear identifiable
from parents to engage in even mundane actions such as picking cause initially, and often it is necessary to rule out possible
up a ball off the ground while playing. His mother reported organic causes of pediatric catatonia. An underlying organic
some “obsessive–compulsive behavior,” with Boy A insisting condition could be identified in ∼20% of pediatric catatonia
that she repeat certain actions and he still showed some motor cases (6, 15). It is important to investigate for these possible
perseveration, such as, sitting slouched with wrists bent but able contributing conditions, as there are specific treatments for
to readjust his position when prompted. He, however, no longer some of them that can improve catatonic symptoms (6, 15).
displayed purposeless movements. The following medical differential diagnoses are important
Lorazepam was consequently increased over a number of to consider: epileptic encephalopathy, infections (e.g., viral
weeks to a daily total of 22 mg in four divided doses, and encephalitis, typhoid fever, toxoplasmosis), autoimmune
fluoxetine increased to 30 mg OD but without any noticeable diseases (e.g., neuropsychiatric systemic lupus erythematosus
change in his depressive symptoms. Our liaison psychiatry and anti-NMDA receptor encephalitis, pediatric autoimmune
colleagues advised a trial of higher doses based on their clinical neuropsychiatric disorders associated with streptococcal
experience of positive response in a subgroup of adolescents. infections (PANDAS), encephalopathy associated with
The decision was made to increase lorazepam to 24 mg daily autoimmune thyroid disease), toxic-induced states (e.g.,
and fluoxetine 40 mg daily, then 50 mg after 1 week, at lithium, ecstasy), and many metabolic and genetic disorders
which there was a remarkable reduction in emotionality and (4, 5, 8, 15).
subsequent complete cessation of tearfulness. Boy A appeared Pediatric catatonia usually has an acute onset, but its onset
more confident, spontaneous in communication and interaction, can also be gradual. Duration can be transient or chronic
and more aware and “awake to his environment.” for weeks or months (4). Children with catatonia are at risk
He equally, remarkably, actively engaged in outdoor activities, of complications secondary to akinesia, such as pneumonia,
including playing table tennis, soccer, and hurling to high malnutrition, dehydration, contractures, decubitus ulcers, or
standards with his parents reporting that he was “like a different thrombosis (4). Catatonia has the potential to progress to
person on the pitch, much more confident and fast.” There was malignant catatonia if left untreated. The use of antipsychotic
no evidence of sedation or other side effects from his medication. medication can increase the risk of progression to malignant
Boy A maintained his recovery on these doses and was able to catatonia (2, 8). Malignant catatonia is a potentially lethal
return to school 2 months after discharge from the hospital, with condition. In this severe form, an exacerbation of motor and
lorazepam dose gradually reduced at the rate of 4 mg every 2 non-motor catatonic symptoms is accompanied by systemic
weeks but maintained on fluoxetine 60 mg daily. symptoms, such as fever, autonomic instability, and delirium
(2, 4, 8).
As in adults, the first-line treatment in pediatric catatonia
DISCUSSION should be benzodiazepines (lorazepam), given IV or
intramuscularly (1, 4, 5, 16). In most cases, symptoms
Schizophrenic disorders are the most common psychiatric improve considerably after lorazepam challenge with 1–
disorders associated with catatonia in children and adolescents 2 mg (4). If a positive response is observed, it is then
(7, 8). Mood disorders are the second most common associated assumed to be catatonia and the dose titrated further for
psychiatric disorder (4). As somewhat evident in this case, optimal response and maintenance of improvement, often
identifiable affective symptoms may only start to emerge after to between 10 and 20 mg per day (4, 5). It is worth noting
the improvement of catatonic symptoms in some cases (4). that the authors made a focused effort to continue with
Considering Boy A’s family history of bipolar affective disorder, lorazepam despite a negative challenge test for reasons
this differential cannot be entirely ruled out, and possibly such a aforementioned and evidence for its effectiveness in the age
diagnosis may become apparent in the future. Traumatic events group. In a prospective study of children and adolescents,
are also important risk factors for the onset of catatonia in benzodiazepines were effective in 65% of 66 patients (16).
children and adolescents (12). This coincides with the theory that The most common side effect observed was excessive sedation
catatonia may represent a primitive evolutionary-based freezing (16). It is also imperative to observe closely for signs of
response to a perceived threat (13). In this case, Boy A’s father’s respiratory depression.
recent hospitalization and psychiatric diagnosis may represent a Although not required in this case, ECT would be the second-
traumatic event for Boy A. line treatment if benzodiazepines were not effective or in severe
Pediatric catatonia is also associated with neurodevelopmental cases such as malignant catatonia (17). The response rate for ECT
disorders. In a review of six studies, an incidence rate of in catatonia in youths is 76–92% (18, 19). However, there can be
4–17% of catatonia was found in adolescents and adults logistic and cultural barriers to using ECT in pediatric patients.
with autistic spectrum disorder (14). Childhood disintegrative Importantly, associated psychiatric or medical conditions
disorders, Tourette’s syndrome, Down’s syndrome, and Prader– may also require separate treatment in addition to the treatment
Willi syndrome are also associated with higher rates of of catatonic symptoms outlined. The authors in this case

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Ridgeway et al. Case Report: Case of Pediatric Catatonia

observed a temporal relationship between administration ETHICS STATEMENT

of high-dose fluoxetine and dramatic improvement in
depressive symptoms. It is however not completely clear Written informed consent was obtained from the minor(s)’
if this effect was synergistic with lorazepam, as existing legal guardian/next of kin for the publication of any potentially
literature appears not to support a role for fluoxetine in the identifiable data included in this article.
treatment of catatonia. Further case reports might be helpful in
this regard. AUTHOR CONTRIBUTIONS
DATA AVAILABILITY STATEMENT LR wrote case report and article. AO was the lead treating
clinician for this case and reviewed the case report and article as
The original contributions presented in the study are included senior author. IM, DD, DK, and ML provided clinical advice to
in the article/Supplementary Material, further inquiries can be AO on the management of the case. All authors contributed to
directed to the corresponding author/s. the article and approved the submitted version.

REFERENCES 12. Dhossche DM, Ross CA, Stoppelbein L. The role of deprivation, abuse, and
trauma in pediatric catatonia, without a clear medical cause. Acta Psychiatr
1. Sorg EM, Chaney-Catchpole M, Hanzen EP. Pediatric catatonia:a case series- Scand. (2012) 125:25–32. doi: 10.1111/j.1600-0447.2011.01779.x
based review of presentation, evaluation, and management. Psychosomatics. 13. Moskowitz AK. “Scared stiff ”: catatonia as an evolutionary-
(2018) 59:531–8. doi: 10.1016/j.psym.2018.05.012 based fear response. Psychol Rev. (2004). 111:984–
2. Cornic F, Consoli A, Tanguy ML, Bonnot O, Perisse D, Tordjman S, et al. 1002. doi: 10.1037/0033-295X.111.4.984
Association of adolescent catatonia with increased mortality and morbidity: 14. Dhossche DM, Shah A, Wing L. Blueprints for the assessment, treatment and
evidence from a prospective follow-up study. Schizophr Res. (2009) 113:233– future study of catatonia in autism spectrum disorders. Int Rev Neurobiol.
40. doi: 10.1016/j.schres.2009.04.021 (2006) 72:267–84. doi: 10.1016/S0074-7742(05)72016-X
3. Ghaziuddin N, Dhossche D, Marcotte K. Retrospective chart review of 15. Lahutte B, Cornic F, Bonnot O, Consoli A, An-Gourfinkel I, Sedel F, et al.
catatonia in child and adolescent psychiatric patients. Acta Psychiatr Scand. Multidisciplinary approach of organic catatonia in children and adolescents
(2012) 125:33–8. doi: 10.1111/j.1600-0447.2011.01778.x may improve treatment decision making. Prog Neuro-Psychopharmacol Biol
4. Benarous X, Raffin M, Ferrafiat V, Consoli A, Cohen D. Catatonia in Psychiatr. (2008) 32:1393–8. doi: 10.1016/j.pnpbp.2008.02.015
children and adolescents: new perspectives. Schizophr Res. (2018) 200:56– 16. Raffin M, Zugaj-Bensaou L, Bodeau N, Milhiet V, Laurent C, Cohen D,
67. doi: 10.1016/j.schres.2017.07.028 et al. Treatment use in a prospective naturalistic cohort, of children and
5. Dhossche DM, Goetz M, Gazdag G, Sienaert P. New DSM-5 category adolescents with catatonia. Eur Child Adolesc Psychiatr. (2015) 24:441–
’unspecified catatonia’ is a boost for pediatric catatonia: review and case 9. doi: 10.1007/s00787-014-0595-y
reports. Neuropsychiatry. (2013) 3:401–10. doi: 10.2217/npy.13.42 17. Cohen D, Paillere-Martinot ML, Basquin M. Use of electroconvulsive therapy
6. Consoli A, Raffin M, Laurent C, Bodeau N, Campion D, Amoura Z, et in adolescents. Convulsive Therapy. (1997) 13:25–31.
al. Medical and developmental risk factors of catatonia in children and 18. Consoli A, Benmiloud M, Wachtel L, Dhossche D, Cohen D, Bonnot O.
adolescents: a prospective case-control study. Schizophr Res. (2012) 137:151– Electroconvulsive therapy in adolescents with the catatonia syndrome: efficacy
8. doi: 10.1016/j.schres.2012.02.012 and ethics. J ECT. (2010) 26:259–65. doi: 10.1097/YCT.0b013e3181fb3924
7. Cohen D, Nicolas JD, Flament MF, Perisse D, Dubos PF, Bonnot O, et 19. Grover S, Malhotra S, Varma S, Chakrabarti S, Avasthi A, Mattoo SK.
al. Clinical relevance of chronic catatonic schizophrenia in children and Electroconvulsive therapy in adolescents: a retrospective study from north
adolescents: evidence from a prospective naturalistic study. Schizophr Res. India. J ECT. (2013) 29:122–6. doi: 10.1097/YCT.0b013e31827e0d22
(2005) 76:301–8. doi: 10.1016/j.schres.2005.01.014
8. Takoaka K, Takata T. Catatonia in childhood and adolescence. Psychiatr Clin Conflict of Interest: The authors declare that the research was conducted in the
Neurosci. (2003) 57:129–37. doi: 10.1046/j.1440-1819.2003.01092.x absence of any commercial or financial relationships that could be construed as a
9. Cohen D, Flament M, Dubos PF, Basquin M. Case series: catatonic syndrome potential conflict of interest.
in young people. J Am Acad Child Adolesc Psychiatry. (1999) 38:1040–
6. doi: 10.1097/00004583-199908000-00021 Copyright © 2021 Ridgeway, Okoye, McClelland, Dhossche, Kutay and Loureiro.
10. American Psychiatric Association. Diagnostic and Statistical Manual of Mental This is an open-access article distributed under the terms of the Creative Commons
Disorders: DSM-5. 5th ed. Washington, DC: American Psychiatric Publishing Attribution License (CC BY). The use, distribution or reproduction in other forums
(2013). doi: 10.1176/appi.books.9780890425596 is permitted, provided the original author(s) and the copyright owner(s) are credited
11. Benarous X, Consoli A, Raffin M, Bodeau N, Giannitelli M, Cohen D, et and that the original publication in this journal is cited, in accordance with accepted
al. Validation of the Pediatric Catatonia Rating Scale (PCRS). Schizophr Res. academic practice. No use, distribution or reproduction is permitted which does not
(2016) 176:378–86. doi: 10.1016/j.schres.2016.06.020 comply with these terms.

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