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REVIEW ARTICLE |
Division of Clinical Neurosciences, School of Molecular and Clinical Medicine, University of Edinburgh, Western General Hospital, Edinburgh
Division of Psychiatry, School of Molecular and Clinical Medicine, University of Edinburgh, Royal Edinburgh Hospital, Edinburgh
Division of Clinical Neurosciences, School of Molecular and Clinical Medicine, University of Edinburgh, Western General Hospital, Edinburgh
Division of Psychiatry, School of Molecular and Clinical Medicine, University of Edinburgh, Royal Edinburgh Hospital, Edinburgh, UK
Correspondence: Dr Jon Stone, Department of Clinical Neurosciences, Western General Hospital, Edinburgh EH4 2XU, UK. Tel: +44 (0)131 537 2911; fax: +44 (0)131 537 1132; e-mail: jstone{at}skull.dcn.ed.ac.uk
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ABSTRACT |
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 TOP ABSTRACT INTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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Aims To determine the frequency of la belle indifférence in studies of patients with conversion symptoms/hysteria and to determine whether it discriminates between conversion symptoms and symptoms attributable to organic disease.
Method A systematic review of all studies published since 1965 that have reported rates of la belle indifférence in patients with conversion symptoms and/or patients with organic disease.
Results A total of 11 studies were eligible for inclusion. The median frequency of la belle indifférence was 21% (range 0–54%) in 356 patients with conversion symptoms, and 29% (range 0–60%) in 157 patients with organic disease.
Conclusions The available evidence does not support the use of la belle indifférence to discriminate between conversion symptoms and symptoms of organic disease. The quality of the published studies is poor, with a lackof operational definitions and masked ratings. La belle indifférence should be abandoned as a clinical sign until both its definition and its utility have been clarified.
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INTRODUCTION |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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METHOD |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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Inclusion criteria
Studies were included only if they met the following criteria.
Data extraction and analysis
All reports were reviewed independently by three investigators (J.S., R.S.
and A.C.). Discrepancies were resolved by a fourth and fifth adjudicator (M.S.
and C.W.). Data were collected on the frequency of la belle
indifférence, the setting of the study, the sampling method, the
symptoms and case definition of the patients and the year of study. We
calculated odds ratios for those studies that included control groups using
Review Manager 4.2.7 for Windows
(http://www.cc-ims.net/RevMan).
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RESULTS |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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Analysis
The results of the systematic review are shown in
Table 1 and
Fig. 1. The median frequency of
la belle indifférence in studies of 356 patients with
conversion symptoms was 21% (range 0–54%). In studies of 157 patients
with organic disease, the median frequency was 29% (range 0–60%). Four
studies included control groups with disease. Analysis of odds ratios
indicated that one controlled study found la belle
indifférence to be significantly more common in hysteria
(Barnert, 1971), whereas the
other three found no significant differences between patients with conversion
symptoms and controls with organic disease
(Raskin et al, 1966;
Weinstein & Lyerly, 1966;
Chabrol et al, 1995).
An additional study of 30 patients with only organic disease (mainly stroke)
reported la belle indifférence in 27%
(Gould et al,
1986).
When studies were ordered by year of publication, no trend towards an increase or decrease in reporting of la belle indifférence over time was apparent. Of the 11 studies, 7 concluded that la belle indifférence was not helpful for differentiating those with conversion symptoms from those with organic disease. The other 4 did not comment on its utility.
The quality of the studies was generally poor. Only 6 studies were clearly of consecutive patients (Table 1) and only 6 studies were prospective (Table 1). The latter is important because a retrospective case-note review is unlikely to be a valid means of determining the presence of a clinical sign. Only 8 studies recorded the actual physical symptoms that led to the diagnosis of conversion symptoms (Table 1). This is also an important limitation, as it is much easier to detect la belle indifférence in a patient with paralysis than in an individual with non-epileptic seizures who is asymptomatic between episodes.
Only 2 studies clearly described what they meant by la belle indifférence (Ebel & Lohmann, 1995) or referenced another description (Gould et al, 1986), and none discussed any of the difficulties in making this judgement (see below). Although 1 study used a system of re-rating to improve the reliability of the clinical diagnosis of la belle indifférence (Barnert, 1971), these data were not presented in the paper. Finally, in none of the studies were the investigators masked to the patient’s diagnosis when assessing whether la belle indifférence was present.
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DISCUSSION |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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Limitations
The conclusions of this review must be qualified by the limitations
inherent in the studies that it included. In addition, there were limitations
in the methodology used for the systematic review. First, we only included
studies that had been published since 1965. To our knowledge, no large
relevant studies were published before that date. Second, the total number of
patients in the review is small. Third, some of the patients who were included
may have been wrongly diagnosed, although this is unlikely to be a major
factor, as a systematic review found the overall rate of misdiagnosis of
conversion disorder to be only 4% since 1970
(Stone et al,
2005).
Meanings of la belle indifférence
The term la belle indifférence seems to have gained
popularity after Freud used it to describe ‘Elizabeth von R’ in
Studies on Hysteria (Breuer &
Freud, 1895). Freud later attributed the term to Charcot
(Freud, 1915), which suggests
that it may have been widely used from the end of the nineteenth century
onwards. Janet (1907) briefly
mentions indifference to both sensory loss and paralysis in his book, The
Major Symptoms of Hysteria, but does not appear to use the term
‘la belle indifférence’. We could not find the
term in any of the other well-known books about hysteria published at that
time, including Charcot’s translated lectures
(Skey, 1867;
Charcot, 1889;
Savill, 1909;
Fox, 1913), although
indifference to areas of anaesthesia on examination was mentioned in many of
those texts (see below). Thus, if the term was used clinically at that time,
it was not deemed sufficiently important to be included in many texts about
hysteria. It appeared with more regularity towards the middle of the twentieth
century, predominantly in the psychoanalytical literature, before it achieved
more widespread usage.
In tracing the history of the term ‘la belle indifférence’, it is clear that it has had more than one meaning since it was first used. We summarise these below.
Hysterical ‘stigmata’ or sensory signs of which the patient is unaware
The commonest description of indifference in the early literature related
to the discovery of sensory signs or ‘stigmata’ of which the
patient was unaware. Janet expressed this common clinical observation as
follows:
This absence of objective disturbances is mostly accompanied by a very curious subjective symptom; namely, the indifference of the patient. When you watch a hysterical patient for the firsttime, or when you study patients coming from the country, who have not yet been examined by specialists, you will find, like ourselves, that, without suffering from it and without suspecting it, they have the deepest and most extensive anaesthesia.... Charcot has often insisted on this point and shown that many patients are much surprised when you reveal to them their insensibility.
Charcot and Janet described ‘hysterical stigmata’ such as hemisensory disturbance, ipsilateral constricted visual fields and reduced hearing, which were characteristically noticed on examination but not reported by the patient (who nevertheless did report other distressing symptoms). Centuries earlier, similar sensory ‘stigmata’ were used as evidence of witchcraft. This clinical phenomenon continues to be seen frequently and is recognised by neurologists as functional or psychogenic (Toth, 2003). However, lack of awareness of sensory disturbance is distinct from the serene indifference to actual disability that is suggested by contemporary descriptions of la belle indifférence.
Conversion of distress
The classic psychoanalytical interpretation of la belle
indifférence is that it is evidence that an intrapsychic conflict
has been converted and kept from its unacceptable conscious expression by the
production of a physical symptom–so-called primary gain. Freud was the
first to admit that this process of conversion was not always complete.
However, when it is present la belle indifférence appears to
represent physical evidence of the conversion process at work, and could be
seen as potent evidence of its truth (one reason, perhaps, why it has been
such a celebrated sign; Abse,
1966). As psychodynamic theory has progressed, more complex
hypotheses have arisen to challenge this rather ‘hydraulic’ model
of conversion (Chodoff, 1954; Greenberg & Mitchell,
1983). For example, Merskey
(1995) suggested that some
patients may simply be relieved that they have escaped a more difficult
problem in their life by becoming ill. However, the simple conversion model is
still the most well known, perhaps because of the persistence of the term
‘conversion disorder’.
The simple conversion hypothesis is at odds with what is known about the frequency of psychiatric disorder and emotional distress in patients with conversion symptoms. Depression and anxiety are reported in 20–50% of patients with conversion symptoms (Wilson-Barnett & Trimble, 1985; Lecompte & Clara, 1987; Crimlisk et al, 1998). In addition, these patients invariably come to medical attention because they are distressed by their symptoms. These observations do not necessarily negate the conversion hypothesis. However, it must now compete with or accommodate other theoretical developments in this area, including the advances in cognitive neuropsychology and neurobiology discussed below (Spence, 1999; Halligan et al, 2001; Brown, 2004).
Alternative explanations for apparent indifference
‘Putting on a brave face’ to avoid a psychiatric diagnosis
Freud’s first use of the term la belle
indifférence–to describe his patient Elizabeth von R in
Studies on Hysteria–implies not so much a denial of disability
that is obvious to everyone else, as ‘putting a brave face on
things’.
She seemed intelligent and mentally normal and bore her troubles, which interfered with her social life and pleasures, with a cheerful air–the belle indifférence of a hysteric, I could not help thinking.
Patients with physical symptoms that cannot be explained by organic disease commonly combine clear distress about their physical symptoms with apparent resilience and cheerfulness. However, such cheerfulness is often easy to expose as superficial and as a ‘mask’ for the depression or anxiety that is identified by a more searching interview. In many cases, strenuous efforts at cheerfulness may simply reflect a desire by patients not to see themselves or be labelled by others as ‘depressed’ or ‘psychiatric’ cases. The following anonymised case from our own recent practice illustrates this:
A young woman had an attack characterised by panic with prominent dissociation, unresponsiveness and limb shaking during venepuncture the day after a surgical procedure. After a period of drowsiness she was found to have a marked right hemiparesis. Investigations to search for a neurological cause of her symptoms were negative and there were positive clinical features in favour of a diagnosis of conversion disorder, including a tubular visual field and strongly positive Hoover’s sign on the affected side. The referring doctors commented on her affect, which was recorded in the notes as ‘unconcerned’, ‘unusually cheerful’and ‘indifferent’. Nursing staff agreed that this was her consistent affect. At interview the patient smiled frequently and did indeed appear unworried by her hemiparesis, even though she had no movement in her right arm and was unable to walk. After 20 min of interviewing, the patient was asked about her apparently cheerful demeanour. ‘Is this really how you are feeling about things or do you think you might be"putting a brave face on things"?’ The patient burst into tears and admitted being terrified both by her symptoms and by the possibility that some one was going to think she had ‘gone crazy’.
Patients often view psychiatric labels for physical symptoms as an implication that the symptoms are fabricated, imagined or relate to ‘going mad’ (Stone et al, 2002a). In addition, patients with conversion symptoms tend to express the conviction that an organic disease is responsible for those symptoms even more strongly than patients whose symptoms are actually a result of an organic disease (Creed et al, 1990; Binzer et al, 1998). It is hardly surprising, therefore, that many patients with these symptoms may try hard not to appear like ‘psychiatric’ cases. Thus, superficial cheerfulness in the face of adversity in an attempt to avoid a psychiatric diagnosis is not the same as indifference to physical disability as implied by la belle indifférence.
Attentional impairment or‘absent-mindedness’
Another difficulty in the assessment of la belle
indifférence is defining for how much of the time the indifference
is present and whether it is present when the patient is specifically asked
about their disability. For example, a patient may appear indifferent most of
the time but be quite clearly concerned when asked about their paralysed
leg.
Lasegue and Janet wrote about the ‘absent-mindedness’ of ‘hystericals’ (Janet, 1901). Lasegue considered it to be a core psychological feature related to ‘general preoccupation’. Janet described it as follows:
an exaggerated state of absent-mindedness, which is not momentary and is not the result of voluntary attention turned in one direction; it is a state of natural and perpetual absent-mindedness which prevents those persons from appreciating any other sensation except the one which for the time occupies their mind.
Such ‘absent-mindedness’ would not be calm acceptance of disability but simply a general diminution of attention masquerading as indifference. The findings of a neuropsychological study of patients with conversion disorder have provided some support for this attentional hypothesis (Roelofs et al, 2003).
La belle indifférence as a marker of factitious disorder
One final possible explanation of la belle indifférence is
that it is the affect of someone who knows that their symptoms are under
conscious control and who is therefore not concerned about them. There are no
data to support or refute this hypothesis.
La belle indifférence: a biological perspective
If we accept that la belle indifférence does sometimes
occur in conversion disorder, are there plausible biological reasons why this
may be so? Anosognosia (denial of hemiplegia) and anosodiaphoria (indifference
to hemiplegia) are surprisingly common clinical features of hemispheric
lesions, particularly right parietal stroke. In one study, anosognosia was
found in 28% and anosodiaphoria in another 27% of 171 patients with right
hemisphere stroke (Stone et al,
1993). Many authors have suggested that this may tell us something
about the biology of la belle indifférence in conversion
disorder. Functional neuroimaging is certainly now being used to explore the
neural correlates of ‘hysterical’ motor and sensory symptoms. For
example, in one study the hypoactivation of the contralateral thalamus seen in
patients with hemisensory conversion symptoms recovered when the symptoms
resolved (Vuilleumier et al,
2001). Perhaps similar dysfunction of parietal areas could lead to
la belle indifférence. However, there are two problems with
this. First, as we have already mentioned, the existence of la belle
indifférence is under threat because of its poor definition and
the potential for misdiagnosis. Second, part of this biological hypothesis of
la belle indifférence has been based on the idea that
conversion symptoms, like neglect, invariably lateralise to the left side of
the body. Both a recent study (Stone
et al, 2002b) and an earlier systematic review
(Jones, 1908) found that there
was little evidence to support this hypothesis, particularly when the symptom
is paralysis.
An alternative but again unproven biological explanation for la belle indifférence is that patients with severe conversion symptoms have frontal hypoactivation (Spence et al, 2000) that could potentially contribute to a syndrome of apathy and indifference.
Other clinical signs of conversion disorder
The survival of la belle indifférence as a clinical sign
over the past century should also be viewed in the context of the other
clinical signs of conversion disorder/hysteria, such as collapsing weakness
and ‘midline splitting’ of sensory loss. These signs have rarely
been assessed in clinical studies and often show poor reliability for the
identification of conversion disorder when they are tested in this way
(Stone et al,
2002c). Although some clinical signs, such as
Hoover’s sign for paralysis, have recently been shown in some small
studies to be potentially more reliable
(Ziv et al, 1998), it
is perhaps not surprising that, among such untested signs, la belle
indifférence has survived unchallenged for so long.
Theoretical and clinical implications
It is not difficult to see why la belle indifférence has
continued to be included as a feature of conversion disorder. First, it has a
romantic history providing a link between modern practice and famous
historical figures such as Charcot and Freud. Giving any clinical sign a
memorable name tends to heighten its profile (and doing so in French perhaps
heightens it even more). Second, it is consistent with beliefs about the
conversion of emotional distress into physical symptoms, which despite the
lack of evidence for them are widely held. Third, theories linking la
belle indifférence to right hemisphere dysfunction may have
promoted the survival of the concept in an era of biological psychiatry.
Fourth, it is yet another untested clinical sign among other untested clinical
signs for ‘hysteria’. Finally, clinicians may not always have
considered the ‘differential diagnosis’ of an apparently
indifferent state. In our experience, this is most commonly manifested as an
apparently cheerful patient with disability who is actually distressed but who
makes strenuous efforts to avoid providing possible evidence for those seeking
to make a psychiatric diagnosis, and thus to avoid the stigma associated with
the latter.
The findings of this systematic review do not support the use of la belle indifférence as a clinical sign for discriminating between conversion symptoms/hysteria and organic disease. The review also highlights the poor quality of the published studies that have addressed the subject, and raises questions about what la belle indifférence actually means. We conclude that further research is required to define and study apparent indifference, in particular looking for alternative explanations for this sign. Despite its attractive name, la belle indifférence should be abandoned as a clinical sign until both its definition and its utility have been clarified.
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Clinical Implications and Limitations |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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LIMITATIONS
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ACKNOWLEDGMENTS |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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REFERENCES |
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TOPABSTRACTINTRODUCTIONMETHODRESULTSDISCUSSIONClinical Implications and...ACKNOWLEDGMENTSREFERENCES |
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Received for publication June 15, 2004. Revision received February 14, 2005. Accepted for publication May 3, 2005.
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Conversion symptoms/hysteria (n=356);
organic disease (n=157).