Copyright ª Blackwell Munksgaard 2004 Bipolar Disorders 2004: 6: 162–165
BIPOLAR DISORDERS
Case Report
Folie a deux in bipolar affective disorder: a case report Patel AS, Arnone D, Ryan W. Folie a deux in bipolar affective disorder: a case report. Bipolar Disord 2004: 6: 162–165. ª Blackwell Munksgaard, 2004 Background: The syndrome of folie a deux is uncommon and often described in the context of schizophrenia. We report a case of induced delusional disorder associated with bipolar affective disorder (BAD). Case report: We present a case of monozygotic twins in their late 60s with an unusually close relation with one another and relative isolation from other people. Both twins have been diagnosed as suffering from BAD and relapsed into mania with psychotic symptoms. During their hospital stay they exhibited features consistent with folie a deux. Separation caused disappearance of the phenomenon whilst the affective disorder persisted. Conclusion: This case highlights the unusual and rare phenomenon of folie a deux occurring in the context of BAD. It also suggests current difficulty in defining folie a deux as an entity according to current diagnostic criteria.
Folie a deux, was a term first coined by Lasegue and Falret in 1873 (1) and denotes transference of delusional ideas from the primary affected individual to one or more others, in close association (2). This phenomenon is also known by different terminology as ÔInduced delusional disorderÔ in the ICD-10 (3), and ÔShared psychotic disorderÕ in the DSM-IV (4). For the purposes of this report we will use the term folie a deux. Current classification systems describe that only the primary suffers from a genuine psychotic disorder and the delusions induced in the secondary, usually disappear when separation occurs. However, if two independent psychotic disorders are present they should be classified under alternative diagnostic categories of the ICD-10 and DSM-IV. This is commonly described in the presence of schizophrenia (5). Only one case report of folie a deux associated with bipolar affective disorder (BAD) is described in the literature (6). We present a case of monozygotic twins who independently presented with
Each author declares that they have no potential conflict of interest.
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Anish S Patela, Danilo Arnoneb and William Ryanc a
Mental Health Unit, Horsham Hospital, St George’s Medical School, Horsham, West Sussex, b Springfield Hospital, St George’s Medical School, London, cSouthlands Hospital, St George’s Medical School, Shoreham, West Sussex, UK
Key words: affective psychosis – bipolar affective disorder – folie a deux – induced delusional disorder – shared delusional disorder – twins Received 11 July 2003, revised and accepted for publication 7 January 2004 Corresponding author: Anish Patel, 11 Onslow Court, Brighton Road, Worthing, West Sussex, BN11 2PL, UK. Fax: 00 44 1903 843889; e-mail:
[email protected]
relapse of BAD with psychotic features, and in the midst of their illness exhibited features consistent with folie a deux. While folie a deux is rare, a clinical presentation of affective disorder in this syndrome is still rare. This mode of presentation of the syndrome we describe is difficult to classify according to the current diagnostic criteria.
Case report
Background history
Twins A and B are 68 years old, monozygotic twins, diagnosed as suffering from BAD according to the ICD-10. Both twins were spinsters and shared a close relationship since childhood and lived in relative social isolation, with twin A being dominant of the two. At the age of 36 years twin A, following a road traffic accident, sustained a severe head injury complicated by a right frontal lobe infarct and epilepsy. She subsequently took early retirement. Twin B shortly after this developed mental health problems, leading to a diagnosis of BAD. She also took early retirement. It was not long after this that
Folie a deux in bipolar affective disorder twin A was also diagnosed as suffering from BAD, which was more severe because of the complicating head injury. Since their illnesses started, both twins have relapsed several times due to non-compliance with medication, but they have never been admitted to hospital simultaneously. Twin A was admitted in August 2002 with a seizure following a chest infection. She had been prescribed lithium in the community but her compliance was poor. When she improved physically, she exhibited confusion and disinhibition. She was consequently admitted to the psychiatric ward. On mental state examination she appeared distractible with pressure of speech. Her mood was elated and her thoughts exhibited flight of ideas with tangential replies. She expressed grandiose delusions that she was pregnant and was going to give birth soon. She denied any perceptual abnormalities. Her cognitive function was difficult to assess fully at the time and she remained without insight. The presentation of twin A was more severe of the two because of her concomitant frontal lobe damage. This had previously left her with personality changes of disinhibition and reduced social control. One week later twin B was assessed in the community and admitted under section 3 of the Mental Health Act 1983 for England and Wales. She was admitted to the same psychiatric hospital but to a different ward. She had been prescribed lithium too, but had poor compliance. Twin B presented unkempt with incoherent speech and mildly elated mood. She exhibited loosening of associations, and expressed grandiose delusions, religious in content. She also experienced auditory hallucinations in the form of classical music playing in the background and lacked insight. Both twins were diagnosed with relapse of their bipolar illness, currently manic with psychotic features. Investigations and treatment
Routine investigations on both twins included baseline blood tests, CT scan and EEG which reported no new findings from previous. Formal cognitive assessment for twin A from a previous consultation was normal. Treatment was initiated for twin A in the form of olanzapine and lithium. For twin B we commenced risperidone and lithium. For both twins benzodiazepines were also used at times of agitation. Progression in hospital
At the time of admission, twin A expressed delusions that she was pregnant and was going to give birth to twins. She appeared to be more elated
and grandiose of the two. During the initial 3-week period, although both twins were on separate wards, regular contact during the day was permitted. During this time twin B began to express very similar delusions that she too was pregnant and was going to give birth to twins. Both twin A and B now shared the same delusion that they were both pregnant with twins, and were supporting and accepting of the fact that each other was also pregnant with twins. After about 3 weeks twin A believed she had given birth to twins, the belief was supported by twin B who also continued with her delusion of being pregnant. The shared delusion persisted for another 2 weeks whilst the twins kept in close contact during the day. However, due to diarrhoea and vomiting outbreak on one of the psychiatric wards, no further contact was permitted between the twins. In a matter of days twin B discarded the belief she was pregnant although she still remained unwell and was still mildly elated. The belief of twin A that she had been pregnant and had given birth persisted until her mental state improved. Following successful treatment, the twins were discharged back to the community with continuing psychiatric support. Discussion
Folie a deux is a rare syndrome which has long attracted clinical attention. We feel this case highlights the following points, which are worthy of further discussion: 1. While folie a deux is rare, a clinical presentation of affective disorder in this syndrome is still rare. 2. The case report draws attention to the fact that the current diagnostic criteria do not acknowledge the possibility of psychiatric morbidity in the ÔassociateÕ. 3. The case offers an opportunity to study the evolution of delusional ideas, in reference to the premorbid relationship between the twins, their mood states and the events that occurred during the admission. A recent review of the literature on folie a deux (5) examined a total of 123 published cases between 1942 and 1993. It found that the diagnosis in the ÔprincipalÕ of an elated mood disorder was 3.7% compared with schizophrenia being 44%. In the ÔassociateÕ 62.5% were found to have psychiatric comorbidity approximately one-third of which was depression. What this data show is firstly that the syndrome of folie a deux, though commonly seen in schizo-
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phrenia is also found to occur in other psychiatric disorders, this is acknowledged in the current diagnostic criteria with reference to the ÔprincipalÕ. However, secondly, the data shows that psychiatric disorders in the ÔassociateÕ are not uncommon, but this is not reflected in the current diagnostic criteria. The relatively rare incidence of this disorder has made it more difficult to determine aetiological factors such as whether social isolation alone in a close relationship is sufficient to precipitate folie a deux. It has been hypothesized that there is an inherited predisposition to an impaired ability to test social reality, taking refuge in fantasies and daydreams (7). Therefore premorbid social isolation and genetic and environmental factors seem relevant in the aetiology as recognized by many authors (8, 9). However, Scharfetter has examined the contribution of schizophrenic hereditary predisposition in folie a deux, independent of environmental influences (10). He concluded that a hereditary predisposition to schizophrenia was required for the development of a folie a deux: Ôonly persons with a genetically determined predisposition are likely to develop a schizophreniform psychosis themselves under the influence of a primary schizophrenic partnerÕ (10). This concept could be translated across to BAD, which is well known to have a significant genetic component with 60–70% concordance rate in monozygotic twins compared with 20% in dizygotic twins (11). Therefore, in this context the disorder may only develop in the presence of genetic predisposition in the ÔassociateÕ to a psychiatric disorder. Folie a deux or its equivalent under other synonyms has been diagnosed for more than 100 years but it still remains an elusive entity, difficult to define. Most branches of medicine, with the gradual accumulation of new information, pass through an ÔidentificationÕ phase when new syndromes are being discovered. A further ÔsynthesizingÕ phase is eventually reached where researchers, equipped with greater knowledge of aetiology, pattern of symptoms and treatment, are able to appreciate the wider implications and ramifications of the problem, as well as uncertainties surrounding the initially identified disease pattern. It is already evident in the literature that disagreement of the key features of folie a deux can be seen (8) and that change in our understanding of the syndrome is occurring. This case also provides a chance to analyse the development and change of delusions in respect to the twinsÕ premorbid history, mood state, and dominance in the relationship. The twins led a very close, sheltered life and never married nor had any
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long-term relationships. Therefore the delusion of becoming pregnant during their elated mood state is of interest. It has been suggested by Beck that grandiose delusions appear to arise from earlier daydreams of glory, serving as a compensation for feelings of loneliness, inadequacy and inferiority (12). Twin A was always noted to be the dominant of the two, her dominance increasing through the frontal lobe damage sustained later in life. So it is of interest to have witnessed the fairly dramatic effects of separation on the delusion held by the ÔassociateÕ twin B, emphasizing the importance and strength of the dynamics held in a relationship to create the phenomena of folie a deux. Conclusion
The current diagnostic criteria in the light of growing reports should be revised to include a broader range of psychiatric conditions and acknowledge the possibility of psychiatric morbidity in the ÔassociateÕ. This could aid clinicians to more in-depth probing of patients and their problems, thus leading to renewed thinking through greater understanding of neuropsychological and neuropathological mechanisms underlying the phenomena than purely a phenomonological approach to the problem. Our case has shown the phenomena of folie a deux occurring between two people with independent relapses of their BAD in the midst of a manic phase of their illness. It has shed some light on the evolution of delusional ideas and the external influences maintaining them. This may help clinicians and researchers in the future to focus on the more fundamental questions of psychopathology and related patterns of neuropsychological function. References 1. Lasegue C, Falret J. La folie a deux. Ann Med Psychol 1877; 18: 321–355. English translation and bibliography by Michaud R. Am J Psychiatry 1964; 21 (Suppl. 4): 1–23. 2. Granlinick A. Folie a deux – the psychosis of association: a review of 103 cases and the entire English literature, with case presentation. Psychiatr Q 1942; 16: 491–520. 3. World Health Organization. The ICD-10 Classification of Mental and Behavioural Disorders. Geneva: WHO, 1992. 4. American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders, 4th edn. Washington DC: American Psychiatric Association, 1994. 5. Silveira JM, Seeman MV. Shared psychotic disorder: a critical review of the literature. Can J Psychiatry 1995; 40: 389–395. 6. Ryan CMD, Khan SA, Warwick HMC, Mindham RHS. Reversal of roles in folie a deux associated with manic depressive illness. Ir J Psychol Med 1992; 9: 55–57.
Folie a deux in bipolar affective disorder 7. Cameron N. The paranoid pseudo-community revisited. Am J Sociol 1959; 65: 52–58. 8. Kallman FJ, Mickey JS. Genetic concepts and folie a deux: a re-examination of Ôinduced insanityÕ in family units. J Hered 1946; 36: 298–306. 9. Soni SD, Rockley GJ. Socio clinical substrates of folie a deux. Br J Psychiatry 1974; 125: 230–235.
10. Scharfetter C. Studies of heredity in symbiontic psychoses. Int J Ment Health 1972; 1: 116–123. 11. Bertelson A, Harvald B, Hauge M. A Danish twin study of manic-depressive disorders. Br J Psychiatry 1977; 130: 330– 351. 12. Beck AT, Rector NA. Delusions: a cognitive perspective. J Cogn Psychother 2002; 16: 455–468.
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