Asylum-seeking children with resignation syndrome: catatonia or traumatic withdrawal syndrome?

Abstract

In the beginning of the 2000s, an increasing number of asylum-seeking children in Sweden fell into a stuporous condition. In the present study, we report 46 consecutive children with the most severe form of this illness where the children were unable to give any response at all, did not react to pain, cold or touching, could not be supported to sit or stand on their feet, could not do anything when requested, and in most cases had enuresis/encopresis. A minority of the children came from war zones (n = 8, 17.4%). A majority belonged to an ethnic or religious minority (n = 32, 69.6%) in their homeland and almost all were persecuted (n = 43, 93.5%). All had either experienced violence themselves or had witnessed or heard about violence against close family members. The age of onset of the first symptom of illness for boys was 11.2 years [CI 9.6–12.8], for girls 11.8 yrs.[CI 10.4–13.2], and the age for falling into stupor for boys was 12.9 years [CI 11.6–14.1] years and was the same for girls, 12.9 years [CI 11.6–14.2] years. Girls tended to have depression before entering the stuporous condition, while the boys tended to have PTSD first (Chi-square = 3.73, p = 0.054). A majority of the children had one (n = 13, 28.3%) or both parents (n = 14, 30.4%) suffering from mental or severe physical disorder. It is discussed whether the presented condition is a separate entity or if the syndrome should be regarded as a variant of catatonia, and whether benzodiazepines should be tried.

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European Child & Adolescent Psychiatry (2020) 29:1103–1109
https://doi.org/10.1007/s00787-019-01427-0
ORIGINAL CONTRIBUTION
Asylum‑seeking children withresignation syndrome: catatonia
ortraumatic withdrawal syndrome?
Anne‑LiisvonKnorring1 · ElisabethHultcrantz2
Received: 12 March 2019 / Accepted: 16 October 2019 / Published online: 1 November 2019
© The Author(s) 2019
Abstract
In the beginning of the 2000s, an increasing number of asylum-seeking children in Sweden fell into a stuporous condition.
In the present study, we report 46 consecutive children with the most severe form of this illness where the children were
unable to give any response at all, did not react to pain, cold or touching, could not be supported to sit or stand on their feet,
could not do anything when requested, and in most cases had enuresis/encopresis. A minority of the children came from war
zones (n = 8, 17.4%). A majority belonged to an ethnic or religious minority (n = 32, 69.6%) in their homeland and almost all
were persecuted (n = 43, 93.5%). All had either experienced violence themselves or had witnessed or heard about violence
against close family members. The age of onset of the first symptom of illness for boys was 11.2years [CI 9.6–12.8], for girls
11.8 yrs.[CI 10.4–13.2], and the age for falling into stupor for boys was 12.9years [CI 11.6–14.1] years and was the same
for girls, 12.9years [CI 11.6–14.2] years. Girls tended to have depression before entering the stuporous condition, while the
boys tended to have PTSD first (Chi-square = 3.73, p = 0.054). A majority of the children had one (n = 13, 28.3%) or both
parents (n = 14, 30.4%) suffering from mental or severe physical disorder. It is discussed whether the presented condition is a
separate entity or if the syndrome should be regarded as a variant of catatonia, and whether benzodiazepines should be tried.
Keywords Resignation syndrome· Traumatic withdrawal syndrome· Catatonia· Asylum seeking· Children·
Hopelessness· Fear
Introduction
In the beginning of this century, an increasing number
of asylum-seeking children in Sweden went into a so far
unknown stuporous condition. This condition was not rec-
ognized by pediatricians and child psychiatrists, although
it had already been described in 1958 by the Swedish child
and adolescent psychiatrist Anna-Lisa Annell as a very rare
disorder occurring mostly after severe psychological trauma
[1]. The Swedish Association of Child and Adolescent Psy-
chiatry screened all child and adolescent psychiatry clin-
ics in Sweden in 2004 and found that 424 refugee children
and adolescents 0–20years had been treated from 2003 to
June 2005 because of reduced communication, motor skills
and ability to carry out daily routines. Approximately, 1 out
of 3 of the 425 were fed by means of a nasogastric tube
as they were unable to eat or drink. Those children who
needed a nasogastric tube were also mute and were laying
down. They were not moving at all, were hypotonic, and
totally blocked from the environment without any formal or
emotional contact with people in their environment. Their
eyes were closed all the time. The children did not react on
touch, sound, pain or cold. Most had enuresis and encopre-
sis. Some parents managed to take them on a wheelchair
regularly to the toilet. Those symptoms were later classified
as symptoms “grade 2” of this unknown disease, which in
the beginning was called “apathy”, and later on when more
knowledge and experience had been gathered: “resignation
syndrome”. If the children could show some response when
spoken to, walk with support, do things when requested and
could be spoon fed, the condition was classified as “grade 1”
[2]. On July 1, 2014, the diagnosis of resignation syndrome
was included in the Swedish version of ICD-10 with classi-
fication, ICD-10-SE; F 32.3A. Since then, this diagnosis has
* Anne-Liis von Knorring
anne-liis.von_knorring@neuro.uu.se
1 Department ofNeuroscience, Child andAdolescent
Psychiatry, Uppsala University, 75185Uppsala, Sweden
2 Division ofORL, Department ofClinical andExperimental
Medicine, Linköping University, Linköping, Sweden
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1104 European Child & Adolescent Psychiatry (2020) 29:1103–1109
1 3
been reported in the register of the National Board of Health
and Welfare, making studies in epidemiology possible.
Very few children with the same or similar symptoms
have been reported from other European countries [3].
Recently, however, there have ben reports from Australia
of a number of refugee and asylum-seeking children with a
syndrome very similar to the resignation syndrome among
those who had been on the Island Nauru for several years
[4].
Aims ofthestudy
The aims of the present study were:
1. to describe the background of the children with a resig-
nation syndrome with regard to traumatic experiences,
parental mental health and family situation;
2. to describe the course of mental illness before the onset
of the resignation syndrome;
3. to discuss the classification of the syndrome.
Methods
46 consecutive children with RS grade 2 (n = 46, 22 boys,
24 girls) were included during the years 2010 to 2018. One
child treated during 2004 at the Department of Child and
Adolescent Psychiatry, Uppsala University Hospital, was
also included. They all had been examined by one or both
of the authors more than once. Three families had two chil-
dren with resignation syndrome. Since both siblings with
resignation syndrome were included in the series, in total six
children had a sibling with the same symptoms. All families
were in the asylum process. For details see Table1.
Examination
All children were examined at least two times. Information
on all previous traumas in homeland, during the escape, and
in Sweden was asked from the parents. As the children did
not communicate, the parents also were systematically asked
about earlier mental and somatic symptoms. Medical records
from general practitioners, pediatric and child psychiatric
departments were studied, as well as medical statements
from physicians and social workers obtained. Diagnoses
according to ICD-10 research criteria of mental disorders
were used [5]. All diagnoses of mental disorders were made
by the same trained, experienced child psychiatrist (ALvK).
The Swedish version of Children’s Global Assessment
Scale (C-GAS) was used to retrospectively estimate the
function of the children at arrival to Sweden and later on at
all examinations [6].
The children’s current loss of functions was described in
terms of capacity to communicate, move, eat and drink and
carry out daily routines. The capacity to carry out any such
tasks was rated on a modified scale ranging from normal (0)
to total loss of function (−4 to −6), meaning that a lower
number indicates a more severe condition. In this scale, the
lowest possible score was −39 [7]
Statistics
In the presentation of the material, descriptive statistical
methods have been used, i.e., mean, range and [CI].
When distributions are compared, the Chi-square test
with one degree of freedom was used. Differences between
means were tested by means of the Mann–Whitney U test
due to non-normal distributions. All analyses were done
using SPSS 24.
Results
Subjects
Table1 demonstrates the demography of the 46 included
children. Eight of them came from ex-Yugoslavia (3 boys,
5 girls), 5 from Iraq/Syria (1 boy, 4 girls), and 33 from ex-
Soviet Union/Russia (18 boys, 15 girls). All children except
one (n = 45) were taken care of by their parent(s) at home
with support from health and social services, after an ini-
tial hospitalization of 3–10days. One child had only been
treated in hospital. They all had the most severe form of
the resignation syndrome (grade 2) and all except two were
tube fed, when examined for the first time. One had been
tube fed prior to our first examination, and the other after.
A minority of the children studied came from war zones. A
majority belonged to an ethnic or religious minority (n = 32,
69.6%) in their homeland, Uighurs (n = 8, 17.4%), Romani
(n = 7, 15.2%), Yezidis (n = 6, 13.0%), Armenians in Russia/
Ukraine (n = 6, 13.0%), and others (n = 5, 10.9%). Almost
all had been persecuted in the homeland. Fourteen (30.4%)
had parents, who fled for political reasons. Only one boy was
unaccompanied by his parents, who probably were dead. Six
Table 1 Demographic information from 46 children with resignation
syndrome
Boys
n (%)
Girls
n (%) p value Total
n (%)
Sex 22 24 n.s 46
Oldest sibling or only
child
16 (72.3) 9 (37.5) 0.02 25 (54.3)
Minority, ethnic, religious 14 (63.6) 18 (75.0) n.s 32 (69. 6)
Denied residency 16 (72.7) 22 (91.7) n.s 38 (82.6)
No final decision 6 2 n.s 8
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1105European Child & Adolescent Psychiatry (2020) 29:1103–1109
1 3
boys came together with only one parent, two fathers and
four mothers. Three of these boys were the only children.
Trauma
Most of the children had in their homeland been forced
to witness violence, rape or killing, and/or threats against
a close family member or had been victims themselves.
Thirty-seven children (80%) had been exposed to such expe-
riences repeatedly. There was no statistically significant dif-
ference between boys and girls with respect to the type of
traumatic event(s). For details see Table2.
Pathogenesis
44 (95.6%) children suffered from posttraumatic stress syn-
drome (PTSD) and/or a depressive episode, which developed
to resignation syndrome. There was a tendency for girls to
have depression before resignation syndrome, while the boys
first had PTSD (chi2 = 3.73, p = 0.054). Three children had
made suicide attempts, and another three had communi-
cated suicidal ideation to their parents. Two children reacted
immediately with severe anxiety after being informed about
deportation by a clerk at the Migration Board and within
a few days both were not eating, moving, hypotonic, and
totally blocked from the environment without any formal or
emotional contact (Table3).
Onset
The two most common first symptoms of resignation syn-
drome the parents reported was fear in 11 cases (8 boys, 3
girls, chi2 = 3.59, p = 0.06) and reduced speech in 11 cases (6
boys, 5 girls, n.s). Sleeping problems were reported in nine
cases (3 boys, 6 girls, n.s), depressive mood in five cases (2
boys, 3 girls, n.s), and withdrawal in five cases (3 boys, 2
girls, n.s). Loss of appetite was reported as the first symptom
in two girls, weakness of the legs in two other girls, and
irritability in one girl.
The age of onset of the first symptom was 11.5years [CI
10.5–12.6], with no significant differences between boys and
girls.
The time from first sign of mental illness to the devel-
opment of the full resignation syndrome varied widely.
The mean time for boys was 682days [CI 230–1128], and
382days [CI 232–531] for girls (n.s). A majority of the
children went from grade 1 to grade 2 of the resignation
syndrome in just a few days.
Four children (3 boys, 1 girl) already suffered from res-
ignation syndrome grade 2, and two children suffered from
RS grade 1 (1 boy, 1 girl) when they arrived at Sweden.
The children with grade 2 had the lowest level of function-
ing on the C-GAS at arrival and those with grade 1 were
assessed with a C-GAS of 25. The C-GAS for all 46 children
at arrival to Sweden (retrospectively assessed) and at our
first examination is shown in Fig.1.
Table 2 Background of trauma
for the 46 asylum-seeking
children with resignation
syndrome
Type of trauma Boys
n = 22 (%)
Girls
n = 24 (%) p value Total
n = 46 (%)
War zone 5 (22.7) 3 (12.5) n.s 8 (17.4)
Persecuted in homeland 20 (90.9) 23 (95.8) n.s 43 (93.5)
Victim of violence 11 (50.0) 14 (58.3) n.s 25 (54.3)
Witnessed violence inflicted on family member 17 (77.3) 20 (83.3) n.s 37 (80.4)
Victim of rape 0 1 n.s 1
Forced to witness mother being raped 5 (22.7) 6 (25.0) n.s 11 (23.9)
Table 3 Diagnoses of mental
disorders according to ICD-10
prior to the onset of resignation
syndrome (retrospectively
evaluated)
Boys
n = 22 (%)
Girls
n = 24 (%)
Total
n = 46 (%)
Depression prior to resignation syndrome 5 (22.7) 12 (50.0) 17 (37.0)
Posttraumatic stress disorder prior to
Resignation syndrome
16 (72.7) 11 (45.8) 27 (58.7)
Secondary depression 8 (36.4) 4 (16.7) 12 (26.1)
Secondary posttraumatic stress disorder 0 4 (16.7) 4
Suicide attempt/communicated suicidal ideation 1 5 (20.8) 6 (13.0)
Severe acute stress reaction prior to
resignation syndrome
112
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1106 European Child & Adolescent Psychiatry (2020) 29:1103–1109
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Family situation
A majority of the children had one (n = 13, 28.3%) or both
parents (n = 14, 30.4%) suffering from mental or severe
physical disorder. For details see Table4.
The 46 children with resignation syndrome had 86 sib-
lings of whom 4 were diseased. Ten (12.2%) of the siblings
suffered from trauma-related mental disorders, 13 (15.9%)
from depression, 6 from resignation syndrome, 4 had a lan-
guage disorder, 1 suffered from separation anxiety disorder,
and 1 from enuresis. In total, there were 35 (42.7%) siblings
with a mental disorder. Three boys were the only child and
came to Sweden with just one parent. One boy was an unac-
companied minor with no siblings.
Trigger
There were different triggers for the children to go into res-
ignation syndrome: Most common was to fall into coma
after having been present at the meeting with the Migration
Board when informed of the negative decision and the com-
ing deportation (every other child) or they had themselves
opened and read the letter (in Swedish) of rejection without
a parent’s support. Nine children (19.6%) had developed
severe mental symptoms immediately after being a victim
of violence in the home country and three children saw their
mother try committing suicide. These children all had a pro-
gression of other symptoms prior to resignation syndrome.
Three girls (all Yezidis), already with negative decisions,
had repeatedly watched YouTube during August 2014 and
later about how the Islamic State treated people/women in
Syria and Iraq with faiths other than Islam. Three other chil-
dren had a sibling (two) or mother (one) with resignation
syndrome, three were victims of violence or harassment in
Sweden, and two had been exposed to a police raid at home
in Sweden as a triggering factor.
Deportation
Three families had earlier applied for asylum in Sweden and
been deported before any child was ill. When these families
returned to Sweden, two children already suffered from res-
ignation syndrome after new traumas. In the third family, the
child fell ill after rejection of their new asylum application
from the Migration Board.
Four other cases were deported to another EU coun-
try according to the Dublin agreement. Two of them were
treated as inpatients in hospital together with one parent,
and both improved. One was sent back to Sweden directly in
connection with the deportation without entering the country
because of the child´s severe illness. The fourth child was
lost to follow-up.
Only one case with resignation syndrome was deported
back to the homeland, where there was no knowledge about
the condition. The father had to feed his sick child through
the tube with baby formula he bought himself both during
hospital stay and later at home. The tube clogged up and
this family returned after 4 months. The child was then
severely dehydrated and in a worse condition than when he
left. Permanent residency permit was obtained after another
7 months and the child started to recover.
Discussion
This is the first prospective study of a large number of chil-
dren with resignation syndrome. This paper deals with the
phase of their disease, when they have been mostly taken
care of by their parents at home, and the whole family had
lived in insecurity, already with negative decisions or wait-
ing for the last decision.
The most important result is the finding that all asylum-
seeking children who have developed resignation syndrome
in Sweden, or were affected when they arrived, have been
exposed to life-threatening traumas in their homeland,
Fig. 1 Children’s Global Assessment Scale of 46 asylum-seeking
children with Resignation Syndrome (RS), evaluated retrospectively
at arrival to Sweden (striped staples) and when ill, at first examina-
tion (filled staples)
Table 4 Health problems of the parents to the children with resigna-
tion syndrome
Mother
n = 46 (%)
Father
n = 46 (%)
Trauma-related mental disorder 7 (15.2) 6 (13.0)
Depression 10 (21.7) 4 (8.7)
Alcoholism/antisocial personality disorder 0 4 (8.7)
Other mental disorder 2 0
Severe physical illness 3 5 (10.9)
Dead 2 5 (10.9)
Total 24 (52.2) 24 (52.2)
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1107European Child & Adolescent Psychiatry (2020) 29:1103–1109
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persecution and violence. Most of them belong to ethnic-
suppressed minorities in their homeland. Since only six
children had a sibling also with resignation syndrome, an
individual vulnerability seems to be present apart from dif-
ficult, traumatic living condition. Almost all had a history
of mental illness, depressive disorder and/or PTSD, which
most often had begun in connection with a specific trauma.
There were no significant gender differences with respect to
the vulnerability to develop resignation syndrome.
The acute onset of the resignation syndrome was most
often triggered by a negative decision from the migration
authorities, when either the child read a negative decision in
a letter (written in Swedish) and had to translate the content
to the parents, and/or the child was required to be present
when the negative decision was orally given to the family. In
both those cases, the child, who usually understood Swedish
best, was the first one to understand the negative decision,
before the translation was made to the parents. Some chil-
dren reacted immediately at that point with vomiting or other
physiological symptoms. As it also was common that both
parents and siblings suffered from mental disorder/distress,
the possibility for support within the family was limited.
The child who fell ill was usually the one who had been
responsible in the family, who often acted as a translator
(the oldest or only son), and/or most often the one who had
been witnessed the most traumatic event in the home country
(rape of mother, torture or killing of father).
The children’s reactions at the triggering moment were
very similar to the described concept of learned helplessness
known in many mammals [8]; when all hope for safety seems
to be lost, in an acute fear/stress situation the individual goes
into a catatonic state which is irreversible without intensive
care. Another well-known concept among both mammals
and birds is the acute fear reaction “freezing” or “play dead
reaction” where the oldpart of the vagus nerve seems to be
involved [9, 10]. The neurophysiological mechanisms behind
RS have to be further studied, which also is planned.
Only a few children in our series came from war zones.
Refugee families from war zones have so far got asylum
in Sweden without extensive delay, and their children can
therefore earlier start recovering from the traumas they have
experienced without any more traumatic triggers.
It has been discussed whether the resignation syndrome is
a separate, new entity, or if the condition should be regarded
as a variant of pervasive refusal syndrome, dissociative stu-
por, depressive stupor or catatonia [11, 12]. According to
the definition of pervasive refusal syndrome by Jaspers:
the patients refuse actively and angry to acts of help and
encouragement, and no other psychiatric condition could
better account for the symptoms [13]. Patients with resig-
nation syndrome are hypotonic, and according to ICD-10,
dissociative stupor includes normal muscle tone, and those
with dissociative stupor also react normally to loud noise
and touch. Children with resignation syndrome do not react
to any sensory stimulation, not even pain, and only a few
refused for a short time actively and angry to acts of help in
the early stage of resignation syndrome.
In the new ICD-11, both dissociative stupor and depres-
sive stupor do not remain as special diagnoses [14]. Instead,
the diagnosis is named catatonia associated with another
mental disorder, as in DSM 5 [15].
No doubt, the children with resignation syndrome fulfill
the DSM 5 criteria for catatonia. They present with 3 of the
12 specified criteria, i.e., stupor (no psychomotor activity,
no reactivity to the environment), mutism (no or minimal
verbal response), and negativism (not responding to external
stimuli or instructions). However, the other nine specified
criteria, catalepsy, waxy flexibility, posturing, mannerism,
stereotypy, agitation, grimacing, echolalia or echopraxia,
were not found in any of the cases or had been preceding
symptoms.
Catatonia as a concept was originally introduced by Kahl-
baum and was at that time seen as a separate entity linked
to manic, depressive and psychotic disorders [16]. Later,
Kræpelin [17] linked catatonia to dementia praecox and
Bleuler described catatonia as a subtype of schizophrenia
[18]. In the earlier versions of the ICD and the DSM clas-
sification systems, catatonia was usually linked to schizo-
phrenia. However, Leonhard clearly linked catatonia also to
affective disorders and anxiety disorders [19]. In 1997, Per-
alta etal. presented data to raise the possibility that catatonia
might be a variant of mood disorder or a distinct entity [20].
This view was later introduced in the DSM 5. The process
has been described by Luchini etal. [21]. It has also been
demonstrated that catatonia is rather common in children
and adolescents [22].
However, even if the resignation syndrome is regarded as
a special variant of catatonia, many questions remain unan-
swered. Even if catatonia is regarded as a separate entity, a
number of medical or neurological disorders may appear
with a similar picture or may have an overlap with catatonia
[23]. All children with resignation syndrome in this study
showed the almost identical clinical picture. The patients
included in the present series were not diagnosed with any
medical or neurological disorders and none of them had any
symptoms or signs of schizophrenia.
It has been argued by Shorter and Fink that catatonia is
associated with fear and alarm, triggered by trauma. It has
been linked to the animal defense of tonic immobility in a
predatory environment [24]. In the children described, affec-
tive, fear, and severe trauma-related disorders are common
[25]. The histories of the children in the present study are in
line with such a view. A similar view has also been taken in
Australia, where a disorder with the same background has
been described and labeled traumatic withdrawal syndrome
[4].
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1108 European Child & Adolescent Psychiatry (2020) 29:1103–1109
1 3
So far, there are no controlled studies of treatment options
of catatonia in children, although prospective case series
have been published. Benzodiazepines relieve the symptoms
of catatonia [26]. It would be of interest to try such a treat-
ment regimen in the asylum-seeking children with resigna-
tion syndrome in a systematic study, especially in the initial
phase when the child is anyhow being treated in hospital.
The present care by the parents at home with support of the
health-care system, but without drugs, was earlier sufficient
as long as the time for decision about permanent safety in
the country was some months away [27]. However, now
when many children have been laying in this condition for
3 years and more, a more active medical treatment policy
is suggested.
Conclusion
Resignation syndrome is always related to earlier severe
traumas leading to PTSD or/and depression and has several
catatonic features [28]. If classified as the retarded type of
catatonia, a treatment trial with benzodiazepine is suggested.
Acknowledgements Open access funding provided by Uppsala Uni-
versity. The authors are extremely grateful to all the families who took
part in this study. We also want to thank Lars von Knorring for help
with analyzing the data, and Ed Paulette for proofreading.
Compliance with ethical standards
Conflict of interest Both authors declare that they have no conflict of
interest.
Ethical standards Lawyers, the asylum-seeking parents themselves
or Doctors of the World, Sweden (Medécines du Monde, Non-Gov-
ernment Organizations) approached us about seeing the children who
were in resignation syndrome grade 2. The study was in accordance
with the ethical standards of the Helsinki Declaration and approved by
the Regional Ethics Board in Uppsala (Reference number 2013/105).
Open Access This article is distributed under the terms of the Crea-
tive Commons Attribution 4.0 International License (http://creat iveco
mmons .org/licen ses/by/4.0/), which permits unrestricted use, distribu-
tion, and reproduction in any medium, provided you give appropriate
credit to the original author(s) and the source, provide a link to the
Creative Commons license, and indicate if changes were made.
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