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Catatonia en 14 A.

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69 views6 pages

Catatonia en 14 A.

caso clinico de catatonia infantil

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Mariano Outes
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Zeitschrift für Kinder- und Jugendpsychiatrie und Psychotherapie, 41 (1), 2013, 69–74

F. Häßler
Z. Kinder-Jugendpsychiatr. et al.: Catatonia
Psychother. in a 14-Year-Old
41 (1) © 2013 Girl with
Verlag Hans Huber, Schizoph
Hogrefe AG , renia
Bern

Kasuistik

A Case of Catatonia in a 14-Year-Old


Girl with Schizophrenia Treated with
http://econtent.hogrefe.com/doi/pdf/10.1024/1422-4917/a000211 - Tuesday, October 04, 2016 8:46:08 PM - IP Address:181.170.215.116

Electroconvulsive Therapy
Frank Häßler1, Olaf Reis1, Steffen Weirich1, Jacqueline Höppner2,
Birgit Pohl3, and Johannes Buchmann1
1
Clinic for Child and Adolescent Psychiatry, Neurology and Psychotherapy, University of Rostock,
Germany, 2Clinic for Psychiatry and Psychotherapy, University of Rostock, Germany,
3
Clinic for Anaesthesiology, University of Rostock, Germany

Abstract. This article presents a case of a 14-year-old female twin with schizophrenia who developed severe catatonia following treatment
with olanzapine. Under a combined treatment with amantadine, electroconvulsive therapy (ECT), and (currently) ziprasidone alone she
improved markedly. Severity and course of catatonia including treatment response were evaluated with the Bush-Francis Catatonia Rating
Scale (BFCRS). This case report emphasizes the benefit of ECT in the treatment of catatonic symptoms in an adolescent patient with
schizophrenic illness.

Keywords: catatonia, schizophrenia, adolescence, ECT

Der Einsatz von EKT bei einer 14-jähigen schizophrenen Patientin mit Katatonie

Zusammenfassung. Es wird über den Fall einer 14jährigen schizophrenen Patientin berichtet, die nach der Behandlung mit Olanzapin
eine schwere Katatonie entwickelte. Unter einer kombinierten Therapie aus Amantadin, Elektrokrampftherapie (EKT) und gegenwärtig
Ziprasidon verbesserte sich ihr Zustand merklich. Schwere und Verlauf der Katatonie wurden mit der Bush-Francis Catatonia Rating
Scale (BFCRS) gemessen. Der Fallbericht betont die Nützlichkeit von EKT bei der Behandlung von katatonen Symptomen bei einer
adoleszenten schizophrenen Patientin.

Schlüsselwörter: Catatonia, Schizophrenie, Adoleszenz, EKT

Introduction in the past (Fink, 2011). The DSM-5 classification ac-


knowledges catatonia as an independent entity for the first
Despite the first description of catatonia by Kahlbaum in time (Fink, Shorter & Taylor, 2010). The current version
1874, our knowledge about the phenomenology, diagnosis, of DSM (DSM-IV) allows for a classification of catatonia
etiology, and treatment of catatonia in childhood and ado- only as derived from affective, other psychiatric, or toxic
lescence is still scarce. One reason for this scarcity is that disorders as well as from organic diseases. Organic diseas-
in the past catatonia was frequently diagnosed under other es as causes of catatonia are mentioned in ICD-10 (F06.1)
labels, such as catalepsy, stupor, and encephalitis lethargica as well.
(Shorter, 2012). For adults, catatonia is a well-described Symptoms of catatonia vary from positive motoric
syndrome that appears to be underdiagnosed (Fink & Tay- symptoms, such as excitement, to negative motoric symp-
lor, 2009). Both classification systems, DSM-IV (295.20) toms, such as immobility or stupor. More than 50% of all
and ICD-10 (F 20.2), acknowledge the association of cata- patients suffering from catatonia display a combination of
tonia with schizophrenia. In the upcoming version of DSM- immobility, mutism, withdrawal, negativism, posturing,
5 the direct tie between catatonia and schizophrenia is to grimacing, and rigidity (Rosebush & Mazurek, 2010). Fre-
be eliminated. The DSM-5 consideration of a fifth digit for quencies of catatonia among young patients admitted to
a catatonia specifier might be an improvement. The addi- psychiatric facilities seem to be lower (0.6–17%) than rates
tion of specifiers of this kind, however, has been criticized found in adults (7.6–38%) (Cornic, Consoli & Cohen,

DOI 10.1024/1422-4917/a000211 Z. Kinder-Jugendpsychiatr. Psychother. 41 (1) © 2013 Verlag Hans Huber, Hogrefe AG, Bern
70 F. Häßler et al.: Catatonia in a 14-Year-Old Girl with Schizophrenia

Table 1
Similarities and differences of catatonia and NMS
Similarities Differences
Catatonia NMS
Motor symptoms hypokinesia/akinesia bizarre additional symptoms: posturing, only hypokinesia, rigidity, no major behav-
automatisms, negativism, rarely hyper- ioral abnormalities
kinesia (stereotypies)
Affective symptoms anxieties strong, intense and uncontrollable anxiety less intense and more controllable anxiety
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Onset slower onset sudden start of mental changes, motor ri-


gidity, fever, and autonomous dysregulation
Pathophysiology of right posterior parietal cortical dysfunction blockade of D-2 receptors in striatum with
motor symptoms consecutive dysregulation of subcortical-
cortical connections in the “motor loop”
Pathophysiology of af- deficits in medial orbitofrontal cortical ac- emotional reaction to akinesia with abnor-
fective symptoms tivity mal subcortical-cortical modulation
Treatment first choice: benzodiaze- third choice – if comorbid with schizophre- no neuroleptics
pines (BZP), e.g., Loraze- nia: neuroleptics, predominantly atypical
pam 1–2 mg up to 20 mg/day neuroleptics while maintaining BZPs
second choice in case of – if comorbid with affective disorders: no
nonresponding to BZPs neuroleptics
or in case of fever more
than 39 °C: ECT

2007). For childhood and adolescence, Wing and Shah drome (NMS) largely resembles catatonia and was exten-
(2000) described five essential features of catatonia (1–5) sively discussed in its relationship to catatonia. The most
and three behavioral abnormalities (6–8): commonly held opinion is that catatonia and NMS are two
1. Increased slowness affecting movement and verbal re- entities on the same spectrum (Fink, 1996; Fink & Taylor,
sponses 2001). There are now five different hypotheses concerning
2. Difficulties in initiating and completing actions the nature of the link between the two diagnoses (Vesperini,
3. Increased reliance on physical or verbal prompting by Papetti & Pringuey, 2010):
others 1. NMS is a drug-induced form of catatonia.
4. Increased passivity and apparent lack of motivation 2. NMS is a drug-induced form of malignant catatonia.
5. Day-night reversal 3. NMS and malignant catatonia are the same.
6. Parkinsonian features 4. Catatonia is a risk factor for NMS.
7. Excitement and agitation 5. NMS is a heterogeneous syndrome that includes both
8. Repetitive, ritualistic behavior. catatonic and noncatatonic responses to antipsychotic
drugs.
To evaluate the severity and course of catatonia, including
treatment response, in adolescents, the Bush-Francis Cata- For a clinician, it becomes difficult to distinguish between
tonia Rating Scale (BFCRS) (Bush, Fink, Petrides, Dowl- these two conditions because many diagnostic criteria
ing & Francis, 1996) is seen as the gold standard (Wong, overlap. In a retrospective investigation of clozapine, cata-
Ungvari, Leung & Tang, 2007). The BFCRS consists of 23 tonia was diagnosed in 7.4% of patients while in a trial of
items with 17 items scored on a scale ranging from 0 to 3 risperidone 5% of patients showed catatonic symptoms
and 6 symptoms being scored as either absent or present. (Ungvari et al., 2010). Both studies did not differentiate
The BFCRS is well established in research and practice between catatonia and NMS, a fact that might explain the
(Takaoka & Takata, 2003). Nevertheless, the definition of high rates of catatonic symptoms observed. About 20 cases
catatonia lacks coherence and clarity. of NMS induced by olanzapine have been published (Sri-
Benzodiazepines are the widely recommended first vastava, Borkar & Chandak, 2009). Confusion rises if ari-
choice of drugs for the treatment of catatonia (Francis, piprazole is recommended for the treatment of catatonia
2010; Ungvari, Caroff & Gerevich, 2010); second and third (Viktor & Tamas, 2010), which is also known to trigger
choices for treatment are described in Table 1. Treatment catatonic symptoms (Shepherd, Garza & De Leon, 2009).
differs in third choices according to the comorbid disorder For the clinician, the question often arises how to differen-
of catatonic symptoms. In case a catatonia is associated tiate between symptoms induced by neuroleptics and non-
with a schizophrenic disorder, a cave has to be avoided, induced symptoms, usually comorbid with an affective or
because neuroleptics themselves have been shown to trig- schizophrenic disorder. Table 1 gives an overview of some
ger catatonia. The so-called neuroleptic malignant syn- characteristics we found in the literature (Lee, 2010; Nort-

Z. Kinder-Jugendpsychiatr. Psychother. 41 (1) © 2013 Verlag Hans Huber, Hogrefe AG, Bern
F. Häßler et al.: Catatonia in a 14-Year-Old Girl with Schizophrenia 71

hoff, 1996, 2002; Rosebush & Mazurek, 2010), which can and imperative acoustic hallucinations. Since then she felt
help to differentiate between the two forms of catatonia. anxious and observed, she shook herself and trembled over
Comprehensive reviews concerning this issue were given her whole body. The patient’s thinking was confused, and
by several authors (Lee, 2010; Takaoka & Takata, 2003; Ung- her thoughts were incoherent. Neurological status, labora-
vari et al., 2010) with varying results. Studies such as the one tory findings, liquor cerebrospinalis, video-EEG, and cra-
by Carroll, Kennedy, and Goforth (2000) failed to discrimi- nial MRI remained normal. All findings failed to provide
nate between catatonia and NMS. Characteristics like those evidence for an organic background to the psychotic symp-
listed in Table 1 might provide a first guideline, although the toms. Developmental disorders were excluded by two ex-
differentiation between drug-induced and noninduced cata- perienced psychiatrists. Under combined treatment with
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tonia remains an open question in our view. behavioral psychotherapy and an oral final dose of 600 mg
In case a catatonia is diagnosed for a patient also suffering quetiapine retard, the symptoms of schizophrenia de-
from schizophrenia, some issues should be considered (Fink creased significantly, allowing the patient to be discharged
et al., 2010; Kruger, Bagby, Hoffler & Braunig, 2003). First, from the hospital.
the association of schizophrenia and catatonia appears only At age 13 psychopathological symptoms slowly re-
to be moderate: Among adult catatonic patients about 28% turned. Especially acoustic hallucinations and symptoms
were also diagnosed with schizophrenia (Caroff, Mann, of anxiety cropped up. At the beginning of 2011, at age
Campbell & Sullivan, 2004). In younger patients this associ- 14, our patient had to be treated again as an inpatient. At
ation appears to be even weaker. In 89 cases of children and this point in time, we were confronted with pronounced
adolescents with catatonia analyzed by Takaoka and Takata mood variations and a distinctive affective instability
(2003), schizophrenia was present in 16.9%. Second, patients with ambitendencies. An increase of the quetiapine dose
with schizophrenic catatonia do not respond well to the treat- (maximum 1200 mg) failed to provide substantial effects.
ment with benzodiazepines. A study by Ungvari et al. (2010) Therefore, we started to change medication to olanzapine
reported response rates of 20–30% for schizophrenic pa- interleavingly. One day after the last application of que-
tients, while more than 80% of catatonic patients suffering tiapine (300 mg) and under 20 mg olanzapine the girl be-
from affective disorders reacted positively to benzodiaze- came suddenly stiff showing a distinctive rigidity of the
pines (Rosebush & Mazurek, 2010). Third, other beneficial extremities and the trunk, also developing a captocormia.
medical treatment options were reported for schizophrenic Our patient showed severe states of uncontrollable anxi-
patients, such as antiepileptic drugs or bromocriptine (dopa- ety, restlessness, drivenness, echolalia, verbigerations,
mine agonist). Amantadine (antagonist of the NMDA type excitement, iterations, stereotypies, and a resistance to
glutamate receptor) was also reported to be beneficial (Lee, passive movements. Her thinking and speaking were
2010; Rosebush & Mazurek, 2010; Takaoka & Takata, 2003). completely confused and incoherent. Eventually, within
In case schizophrenic children or adolescents do not 2 days, she developed a catatonic stupor with a waxy
respond to medical treatment, or show a refractory to psy- flexibility (flexibilitas cerea). The creatinine kinase (CK)
chotropic medications, electroconvulsive therapy (ECT) increased within these 2 days to 1071 U/l (reference <
is considered to be an effective treatment alternative (Ta- 109 U/l), myoglobine increased to 171 ng/ml (reference
kaoka & Takata, 2003; Wachtel, Dhossche & Kellner, 25–58 ng/ml). Other laboratory findings remained un-
2011). Recently, an overview was published regarding re- changed (liquor cerebrospinalis, parameters of coagula-
sponses to ECT in catatonic adult patients (Caroff, Ung- tion and inflammation, liver enzymes, potassium, sodi-
vari, Bhati, Datto & O’Reardon, 2007). The authors con- um, chloride), just as did temperature, blood pressure,
cluded that acute hypokinetic catatonia is extremely sen- respiratory rate, diaphoresis, and other vegetative param-
sitive to ECT, achieving response rates of 67% to 100%. eters. However, a constant trembling of the whole body
Sienaert, Vansteelandt, Demyttenaere, and Peuskens was remarkable. A repetition of EEG and cMRI did not
(2010) found no evidence of a deleterious effect of ultra- provide any new results. An intravasal application of bi-
brief bifrontal or unilateral ECT on cognitive function- peridene to evaluate extrapyramidal side effects provided
ing. A study by Rey and Walter (1997) on children and no results. At this time, the hypothesis of an NMS was
adolescents found overall efficacy rates of 80% for ECT. built, despite the fact that an increase of the CK does not
For ECT in younger patients it is important to recognize provide a valid indicator (Lee, 2010). With the differen-
that, compared to adults, children show lower seizure tial diagnosis of a NMS, all neuroleptics were removed
thresholds while ECT seizures last longer (Rey & Walter,
1997). Table 2
Sum scores of the BFRCS during the catatonic stupor state
of the girl, starting with the first day of the rigidity (minimal
0, maximum 66); b = biperidene, d = diazepam, l = loraze-
Case Report pam, h = haloperidol
Day 1 2 (b) 3 (d) 5 (l) 7 (l + h) 10 12
The patient we report about is a 14-year-old female twin
(IQ 53) who at age 12 started to experience visual, haptic, BFRCS 30 42 45 51 40 45 48

Z. Kinder-Jugendpsychiatr. Psychother. 41 (1) © 2013 Verlag Hans Huber, Hogrefe AG, Bern
72 F. Häßler et al.: Catatonia in a 14-Year-Old Girl with Schizophrenia

Table 3
Course during treatment with ECT
Date ECT bitemporal Epileptic sei- Bush-Francis Catatonia Remarks/medication
zure/duration Rating Scale

08.02.2011 Energy set 10% 84 s 37 Lorazepam 2 × 2 mg


Charge delivered 49.3 mC Haloperidol 3 × 5 mg i.v., Clexane 20 mg s.c.
Current 0. 90 A
Stimulus duration 5.6 s
Pulse width 0.50 ms
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Frequency 10 Hz

09.02.2011 No changes 121 s 36 No changes

10.02.2011 No changes 29 s 37 + Antibiotics and Iron

11.02.2011 No changes 20 s 35 Moving to pediatric emergency unit, Amanta-


dine 600 mg/day

14.02.2011 Energy set 15% 250 s 36 Amantadine 900 mg/day


Charge delivered 75.4 mC
Current 0.90 A
Stimulus duration 5.6 s
Pulse width 0.50 ms
Frequency 20 Hz

16.02.2011 No changes 108 s 26 No changes

18.02.2011 No changes 152 s 24 No changes

21.02.2011 No changes 210 s 25 No changes

23.02.2011 No changes 145 s 21 No changes

25.02.2011 Energy set 10% 51 s No changes


Charge delivered 49.3 mC
Current 0. 90 A
Stimulus duration 5.6 s
Pulse width 0.25 ms
Frequency 10 Hz

01.03.2011 No changes 32 s 19 Lorazepam 3 × 0.5 mg


Amantadine 2× 200 mg/day
In days with ECT without morning doses of any
drugs.

04.03.2011 No changes 54 s 19 No changes

08.03.2011 No changes 32 s 8 No changes in medication, transfer back to the


clinic for child and adolescent neuropsychiatry

11.03.2011 Energy set 15% 97 s 12 No changes


Charge delivered 75.4 mC
Current 0. 90 A
Stimulus duration 5.6 s
Pulse width 0.50 ms
Frequency 20 Hz

16.03.2011 Energy set 15% 183 12 Lorazepam 3 × 0.5 mg, Ziprasidone 2 ×


Charge delivered 75.4 mC 20 mg/day on days with ECT without morning
Current 0. 90 A doses.
Stimulus duration 4.2 s
Pulse width 0.50 ms
Frequency 20 Hz

23.03.2011 No changes 222 9 Lorazepam 3 × 0.5 mg, Ziprasidone 60 mg/day,


Amantadine 2 × 200 mg/day
on days with ECT without morning doses of
any drugs.

Z. Kinder-Jugendpsychiatr. Psychother. 41 (1) © 2013 Verlag Hans Huber, Hogrefe AG, Bern
F. Häßler et al.: Catatonia in a 14-Year-Old Girl with Schizophrenia 73

and the girl was treated solely with benzodiazepines (3 × Discussion


10 mg diazepam). Within two days, the CK returned to a
normal degree, but the catatonic stupor and a significant
The patient reported here was a female adolescent with
impairment of vigilance and communication abilities
mild mental retardation who exhibited severe catatonia.
with a pronounced staring remained. At this point, the
She was successfully treated with amantadine HCL, ECT,
diagnosis was rechanged to catatonic schizophrenic stu-
and is currently being treated solely with ziprasidone. Im-
por because catatonia had started as a mix of behavioral
provement was noted after 12 ECT episodes. In sum, she
symptoms, not only as hypokinesia (see Table 1). Neither
received a total of 16 ECT episodes. Our case report is in
diazepam nor lorazepam (3 × 2 mg) improved the status
line with the investigation by Girish and Gill (2003). In
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markedly. Because of the primary schizophrenia we start- their study 14 patients with nonaffective, nonorganic cata-
ed a reattempt with haloperidol (3 × 10 mg) as an ultima tonia who did not respond to benzodiazepines improved
ratio, but failed again. Table 2 shows the daily total sco- rapidly under ECT. Catatonic symptoms in this study, how-
res of the BFCRS during this time. ever, disappeared more quickly than in our case. We ob-
On day 16 after the beginning of the rigidity, we com- served a catatonic status with a prominent stupor combined
menced bitemporal ECT (Thymatron® System IV with with waxy flexibility persisting over 7 weeks. Alterations
GenieTM IV EEG analysis software) with a cluster of dai- of excitement were first seen after the 12th ECT episode.
ly treatments over 4 days. To reduce side effects, espe- With regard to the young age of our patient initial seizure
cially cognitive dysfunctions, we used ECT treatment thresholds (IST) were set at a lower level (49.3 mC and
techniques that in adults have proved to result in smaller 75.4 mC), compared to IST in bilateral ECT in adults
short- or long-term memory deficits (Sackeim et al., (111.6 mC) as reported by Waarde, Verwey, and Mast
2007). In particular, we stimulated her with brief pulse (2009). The treatment with antipsychotics is in line with
form. An individual electrical dosage titration was done more recent strategies reported by Caroff et al. (2007) and
at the beginning of the first ECT session. There were no Rosebush and Mazurek (2010). However, the role of anti-
major effects, but an acute exacerbation of schizophrenic psychotic drugs in catatonia remains unclear to date and
symptoms was seen at the beginning of ECT. For that has been studied insufficiently. We argue here that antipsy-
reason, we started with an accompanying amantadine chotics may be effective in some cases. They should, how-
therapy (3 × 200 mg). With this add-on we aimed at a ever, be handled with greatest care because they may re-
beneficial effect as reported by Carroll, Goforth, and challenge catatonic episodes (Lee, 2010). We agree with
Thomas (2007). After the patient started to show signs of Francis (2010), who proposed a cautious trial of these
malnutrition and a beginning pneumonia with light to agents and careful monitoring for catatonic symptoms.
moderate febrile temperature, she was transferred tempo- Unfortunately, schizophrenic catatonia proves to be a se-
rarily to a pediatric emergency unit where antibiotics vere condition in young people, with 10% mortality at 4-
were added to the treatment. No autonomic abnormalities year follow-up (Cornic et al., 2007), caused by either sui-
appeared further on. cide (n = 2) or by medical conditions (n = 1). In other
Bitemporal ECT was continued three times a week for words, the course of our patient remains uncertain, despite
the next six treatment episodes, then twice a week for the marked reduction of psychopathology and catatonic
another four treatment episodes, and finally once per symptoms. For severe cases of schizophrenic catatonic stu-
week for two maintaining treatment episodes. Lorazepam por refractory to pharmacological treatment, we suggest an
and amantadine treatment was continued over this time intensive and, if necessary, long-lasting ECT sequence –
period with the patient showing slow improvement of the even in adolescents. In our case we saw no memory impair-
motor and the psychopathological state. After 12 treat- ment as measured with the Working Memory Index of the
ments, the girl showed significant improvement; she Wechsler Scales for children. In sum, we regard ECT as a
started to move (answered with little movement or tone) valuable second-choice treatment option (see Table 1) in
and became more verbal. ECT was finished after 16 treat- adolescents with schizophrenic catatonic stupor.
ments. Catatonic symptoms were no longer evident,
though the visual, haptic, and imperative acoustic hallu-
cinations returned, again associated with marked fear.
After careful consideration we decided to start again with References
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Initial Date Submitted January 23. 2012
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sentation, response to benzodiazepines, and relationship to
Neuroleptic Malignant Syndrome. Journal of Clinical Psycho- Disclosures of interest of Dr. Häßler concern the following
pharmacology, 30, 3–10. positions: Advisory Board for Eli Lilly GmbH Germany,
Northoff, G. (1996). Neuroleptic malignant syndrome and cata- Janssen-Cilag, Research Support from Novartis Pharma-
tonia: One entity or two? Biological Psychiatry, 40, 431–432. ceuticals, Bayer Vital, Travel Grants from Novartis Phar-
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