Medical and developmental risk factors of catatonia in children and adolescents: A prospective case–control study

https://doi.org/10.1016/j.schres.2012.02.012Get rights and content

Abstract

Context

Rare diseases have been associated with more and more genetic and non genetic causes and risk factors. But this has not been systematically assessed in catatonia, one of the psychiatric syndromes, that is most frequently associated with medical condition.

Objective

We sought to assess the medical and developmental risk factors of catatonia in children and adolescents.

Methods

From 1993 to 2009, 58 youths aged 10 to 18 years were prospectively admitted for catatonia and were followed up after discharge. A multidisciplinary approach assessed patients' medical condition and developmental history. A causality assessment scored medical risk (maximum score = 10; κ = 0.91). We compared the prevalence of catatonia in these patients to that of 80 inpatients with bipolar I disorder admitted from 1993 to 2003 who were also followed up.

Results

We found that 13 (22.4%) patients had medical conditions and 18 (31%) had a history of developmental disorder in the catatonia group, whereas 1 (1.3%) and 17 (22.6%) patients had the same conditions in the bipolar group (p < 0.001; p = 0.17, respectively). Medical conditions associated with catatonia included auto-immune encephalitis (systemic lupus erythematosus [N = 3] and anti-NMDA-receptor encephalitis [N = 1]), seizures (N = 1), ciclosporin encephalitis (N = 1), post hypoglycaemic coma encephalitis (N = 1), and genetic or metabolic conditions (chorea [N = 2], 5HT cerebrospinal fluid deficit [N = 1], storage disease [N = 1], fatal familial insomnia [FFI; N = 1], and PRODH mutations [N = 1]). Six patients responded to a specific treatment approach related to their medical condition (e.g., plasma exchange in the case of auto-immune encephalitis).

Conclusion

Catatonia in children and adolescents is associated with a high prevalence of medical conditions. This needs to be acknowledged as it may greatly delay the treatment of catatonia and the diagnosis of medically related catatonia. Tragically, this may deny patients treatment opportunities.

Introduction

Catatonia is among the most severe psychiatric syndrome. The prevalence of catatonia in adult inpatients ranges from 7.6% to 38%. Catatonia is more frequent among women and its most commonly associated psychiatric diagnosis is mood disorder (Taylor and Fink, 2003). Catatonia has not been fully investigated in children and adolescents, although it can occur in children and adolescents. In this age group, catatonia is rare, and it increases the risk of premature death (including suicide) by 60-fold, making it the most severe psychiatric condition (Cornic et al., 2009). The prevalence of catatonia in children and adolescent inpatients is estimated to range from 0.6% to 17.7% according to different inclusion criteria (e.g., general psychiatric unit vs. adolescents receiving electroconvulsive therapy (ECT); high income vs. low income country inpatient unit) (Cohen et al., 2005). In contrast to adult catatonia, catatonia in children and adolescents is more frequent in boys than in girls (Cohen et al., 2005, Thakur et al., 2003). The most commonly underlying psychiatric disorder is schizophrenia (Cohen et al., 2005, Takaoka and Takata, 2003), but it can also occur in youths with a history of developmental disorder (e.g., pervasive developmental disorder [PDD] or intellectual disability [ID]) (Dhossche et al., 2006, Wing and Shah, 2000). Gender difference between paediatric and adult catatonia can be explained by gender differences in underlying psychiatric disorders (early onset schizophrenia is more frequent in boys and mood disorders are more frequent in women). However, the clinical presentation of paediatric catatonia is similar to that in adults, which involves psychological symptoms (e.g., mutism and social withdrawal) and motor symptoms (e.g., stupor and waxy flexibility). These symptoms can be continuous or discontinuous. Symptomatic treatments are also similar to that in adult catatonia. The first line of treatment is high doses of benzodiazepines (e.g., lorazepam  15 mg/day). In cases of resistant or malignant catatonia, electroconvulsive therapy (ECT) is recommended, even in patients with PPD or ID (Taylor and Fink, 2003, Wachtel et al., 2008). In addition, clinical practice depends on the possibility of curing the underlying condition. Indeed, catatonia may occur in patients with various psychiatric disorders (usually schizophrenia and severe mood disorders) and medical conditions (e.g., neurological conditions, intoxication, auto-immune diseases, and metabolic conditions) (Cottencin et al., 2007, Lahutte et al., 2008). Some of these conditions have poor prognoses and may result in death (Dimitri et al., 2006). Recognising underlying medical conditions is essential because it may lead to a cure. Given the variety of possible medical conditions, guidelines have been proposed to help clinicians challenged by this complex issue (Lahutte et al., 2008).

Since 1993, we prospectively followed up a sample of youths with catatonia to investigate their socio-demographic characteristics, symptomatologies, psychiatric and medically associated conditions, developmental histories and outcomes (Cohen et al., 1999, Cohen et al., 2005, Cornic et al., 2009). This study aimed to: (1) assess the prevalence of medical risk factors and developmental histories in this sample of patients with catatonia; (2) compare it with the prevalence of medical risk factors and developmental histories found in a sample of inpatients with bipolar I disorder; and (3) assess whether socio-demographic or clinical characteristics help distinguish catatonia associated with medical/developmental risk factors from catatonia with no such risks.

Section snippets

Patient recruitment

We assessed all patients admitted to the Department of Child and Adolescent Psychiatry at University Hospital La Pitié-Salpêtrière between 1993 and 2009 for catatonia. In all, we hospitalised 5532 patients aged 4 to 18 years. To obtain a diagnosis of catatonia, patients must present at least two motor symptoms or one motor symptom and a non-motor symptom indicative of severe behavioural and emotional impairment. The catatonia symptom list (see Supplementary Table 1) was based on the modified

The prevalence of medical risk factors in youths with catatonia

We prospectively included 58 patients with catatonia in the 1993 to 2009 cohort. Table 1 summarises the socio-demographic characteristics. Mean age at admission was 15 (± 1.8) years [range: 9–18]. The sex ratio was 2 males for 1 female. All but 3 patients were post-pubescent. SES varied greatly among participants; 60.3% of the patients were off high and middle SES. There were no significant demographic differences among the BP sample except for age and sex ratio. Patients with catatonia were

Discussion

Considering that this sample was based on young patients who were consecutively hospitalised in a psychiatric ward due to catatonia, the most important result is that 22.4% of the patients presented a medical condition. This risk was significantly higher compared to a sample of inpatients with BP. In contrast, a high prevalence of developmental disorders was also found in patients with BP. This result corroborates a previous review specifying the kinds of medical conditions that are associated

Conclusion

Catatonia in children and adolescents is associated with a high prevalence of medical conditions. This needs to be acknowledged as it may greatly delay the treatment of catatonia and the diagnosis of medically related catatonia. Tragically, this may deny patients' treatment opportunities.

The following are the supplementary materials related to this article.

. Bush and Francis catatonia rating scale modified for use in child and adolescent practice (Bush et al., 1996, Cohen et al., 2005).

Role of funding source

Funding/support: Grants from the French Ministry of Health (Programme Hospitalier de Recherche Clinique AOM 06-088) and the Fondation Wyeth pour la Santé de l'Enfant et de l'Adolescent funded this research. The funding agencies were not involved in the study design, collection, analysis and interpretation of data, writing of the paper, and/or the decision to submit for publication.

Role of the sponsors: None of these non-commercial funding organisations had any role in the design or conduct of

Contributors

Study concept and design: Cohen, Consoli, Bonnot, Laurent.

Acquisition of data: Cohen, Bonnot, Consoli, Amoura, An-Gourfinkel, Sedel, Campion.

Statistical analysis: Bodeau, Cohen.

Interpretation of data: All authors.

Drafting the manuscript: Cohen, Consoli, Bonnot, Laurent.

Critical revision of the manuscript for important intellectual content: Amoura, An-Gourfinkel, Sedel, Campion.

Final draft: All authors.

Conflict of interest

Dr. Bonnot reported receiving travel support from Actelion; Dr. Cohen reported past consultation for or the receipt of honoraria from Schering-Plough, Bristol-Myers Squibb, Otsuka, Janssen, and Sanofi-Aventis; Dr. Consoli reported receiving travel support from Bristol-Myers Squibb; Drs. Amoura, Bodeau, Campion, Laurent, An-Gourfinkel, and Sedel have no relationship that might have interest in the submitted work; their spouses, partners or children have no financial relationship that may be

Acknowledgements

The authors thank all the patients and their families for participating despite invaliding conditions. They also thank Prof. Doug Levinson for careful editing and comments of the manuscript. Marie Raffin and Nicolas Bodeau had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.

References (56)

  • W.H. Green et al.

    Schizophrenia with childhood onset: a phenomenological study of 38 cases

    J. Am. Acad. Child Adolesc. Psychiatry

    (1992)
  • B. Lahutte et al.

    Multidisciplinary approach of organic catatonia in children and adolescents may improve treatment decision making

    Prog. Neuropsychopharmacol. Biol. Psychiatry

    (2008)
  • E. Louet et al.

    Psychodynamic-oriented psychological assessment predicts evolution to schizophrenia at 8-year follow-up in adolescents hospitalized for a manic/mixed episode: interest of an overall subjective rating

    J. Physiol. Paris

    (2010)
  • G. Masi et al.

    Acute catatonia after a single dose of ecstasy

    J. Am. Acad. Child Adolesc. Psychiatry

    (2002)
  • D. Perisse et al.

    Case study: effectiveness of plasma exchange in an adolescent with systemic lupus erythematosus and catatonia

    J. Am. Acad. Child Adolesc. Psychiatry

    (2003)
  • O. Taieb et al.

    Clinical relevance of electroconvulsive therapy (ECT) in adolescents with severe mood disorder: evidence from a follow-up study

    Eur. Psychiatry

    (2002)
  • F.O. Walker

    Huntington's disease

    Lancet

    (2007)
  • M. Zeidler et al.

    New variant Creutzfeldt–Jakob disease: psychiatric features

    Lancet

    (1997)
  • B. Begaud et al.

    Imputation of the unexpected or toxic effects of drugs. Actualization of the method used in France

    Therapie

    (1985)
  • W.R. Breakey et al.

    Typhoid catatonia responsive to ECT

    Br. Med. J.

    (1977)
  • J. Brunelle et al.

    Phenomenology, socio-demographic factors and outcome upon discharge of manic and mixed episodes in hospitalized adolescents: a chart review

    Eur. Child Adolesc. Psychiatry

    (2009)
  • G. Bush et al.

    Catatonia. I. Rating scale and standardized examination

    Acta Psychiatr. Scand.

    (1996)
  • A. Consoli et al.

    Electroconvulsive therapy in adolescents with the catatonia syndrome: efficacy and ethics

    J. ECT

    (2010)
  • A. Consoli et al.

    Malignant catatonia due to anti-NMDA-receptor encephalitis in a 17-year-old girl: case report

    Child Adolesc. Psychiatr. Ment. Health

    (2011)
  • F. Cornic et al.

    Catatonia in children and adolescents

    Psychiatr. Ann.

    (2007)
  • J. Dalmau et al.

    Limbic encephalitis: the new cell membrane antigens and a proposal of clinical-immunological classification with therapeutic implications

    Neurologia

    (2007)
  • M. de Chaldee et al.

    Linkage disequilibrium on the COMT gene in French schizophrenics and controls

    Am. J. Med. Genet.

    (1999)
  • E. De Grandis et al.

    Cerebrospinal fluid alterations of the serotonin product, 5-hydroxyindolacetic acid, in neurological disorders

    J Inherit Metab Dis

    (2010)
  • Cited by (0)

    View full text