Syndrome of subjective doubles as a rare presentation of a first-episode psychosis
- Joao Luis Martins Quarenta 1,
- Cláudia Mota Pinto 2,
- Catarina Pedro Fernandes 3 and
- Pedro Morgado 3 , 4 , 5
- 1 Psychiatry, Hospital Padre Américo Vale do Sousa SA, Penafiel, Portugal
- 2 Psychiatry, Hospital Beatriz Angelo, Loures, Lisboa, Portugal
- 3 Psychiatry, Hospital de Braga, Braga, Braga, Portugal
- 4 Life and Health Sciences Research Institute (ICVS), School of Medicine, Universidade do Minho, Braga, Braga, Portugal
- 5 ICVS/3B’s, PT Government Associate Laboratory, Braga/Guimarães, Portugal
- Correspondence to Professor Pedro Morgado; pedromorgado@med.uminho.pt
Abstract
In this paper, we report the case of a man in his 30s with a first-episode psychosis, characterised by a subtype of delusional misidentification syndrome in which the delusion of doubles is exclusively of the patient’s own self. This subdivision can be termed ‘syndrome of doubles of the self’ or ‘syndrome of subjective doubles’. Additionally, an examination of the patient’s mental state showed paranoid delusions. After being evaluated in the emergency department, the patient was hospitalised, and medicated with antipsychotic drugs. One week later, he was discharged with total remission of psychotic symptoms. This is a rare and curious presentation of a psychotic episode, with very few similar cases described to date.
Background
Delusional misidentification syndromes (DMS) are uncommon neuropsychiatric disorders that can be defined by a set of rare delusions, in which there is a fixed false belief about the identity of a significant person like a family member or oneself, place or object and are characterised by face recognition memory impairments.1 2
These delusional syndromes are often accompanied by paranoid delusions and are not diagnostic entities per se.3
Originally, this psychopathological phenomenon was only linked to psychiatric diseases. Some authors considered DMS to be one of the most persistent psychotic symptoms in patients with schizophrenia. However, with the ever-evolving neurosciences domain and contribution from other medical specialties, such as neurology and neuroradiology (in the study of its aetiology), it is known that this phenomenon can occur in several neuropsychiatric disorders and it is accompanied by neurobiological findings that support a structural basis to DMS.4–6
These psychotic symptoms were first described by Karl Jaspers within the Delusions of Transformation (Allgemeine Psychopathologie, 1913).7 A few decades later, in 1981, Christodoulou and Malliara-Loulakaki introduced the term ‘delusion misidentification syndromes’. This group of false recognitions shared a common basis and were divided into four main subtypes: Capgras syndrome (CS), Fregoli syndrome, intermetamorphosis and subjective doubles.8
The delusion of doubles exclusively of the patient’s own self is known as the ‘syndrome of subjective doubles’ (Doppelganger), or ‘syndrome of doubles of the self’. In this syndrome, described by Christodoulou in 1978, patients believe that other people become a physical duplicate of themselves, and carry out independent actions.5
It is considered a rare phenomenon, with very few reported cases since its first description, and, within the delusional identification syndromes, is regarded as the rarest of them all.9 The short duration of misidentification syndromes in some patients may be a contributing factor to its underdiagnosis in cross-sectional studies.10
In this article, we describe the case of a patient with the syndrome of subjective doubles during first-episode psychosis. The authors intend to highlight its unique features and shed some light on this rare psychopathological phenomenon.
Case presentation
A man in his 30s with no psychiatric history was taken by a relative to the emergency psychiatry department. He was living with his family at the time and was running his own business. He had no significant medical history except for a psoriasis diagnosis and there was no family history of psychiatric illness. He reported daily cannabis consumption over a 10-year period, but did not meet criteria for DSM-V Substance Use Disorder.
A feeling that something was not quite right started approximately 3 years earlier, after a friend performed a witchcraft ritual on him. He started to exhibit signs of paranoid ideation. At work, he held a firm belief that many of his customers had something to tell him. He also believed that he could influence the results of live sport matches he watched. All of this because the witchcraft ritual was controlling him. He went on to say that a second version of himself, one that was identical in every way, had duplicated his identity 3 years earlier. Two days prior to admission, he described feeling peaceful because everything finally made sense: a mobile phone that looked exactly like his own meant that a second version of himself really existed. It was at this point when he decided to close his business and change the name used for his business affairs.
Collateral information from the patient’s family corroborated that he had been engaging in ruminative, persecutory thought patterns for a few months at least and denied any perceptible abnormality in his mood or sleep patterns.
At the time of his initial psychiatric interview, he maintained eye contact, with no psychomotor agitation or retardation. Attention, concentration and orientation were intact. He described his mood as ‘peaceful’, and his affect was congruent with his mood. His speech was noted to be slightly increased in rate, but easily interruptible when necessary. He exhibited a tangential thought process with content in line with persecutory and self-reference delusions. He did not appear to respond internal stimuli. At the time of the initial assessment, he denied any suicidality or homicidality.
Investigations
Although DMS were initially described in psychiatric patients, such presentations have been increasingly reported in patients with focal neurological lesions.
Full blood count, electrolytes, liver function tests, fasting glucose, cholesterol, triglycerides, endocrine function tests, hepatitis screen, HIV, screening for sexually transmitted diseases, vitamin measurements and ECG showed no abnormalities, except for a mild folic acid deficiency.
Urine drug screening revealed the presence of tetrahydrocannabinol (THC). Physical examination, including neurological examination, revealed no pathological findings. A CT scan of the brain exhibited standard results for the age group. Therefore, MRI was not performed. Investigations were conducted according to box 1.11
Recommended workup for neuropsychiatric conditions associated with delusional misidentification syndromes (DMS)
Broad screening and medical baseline
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Chemistry panel
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Complete blood count with differential
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Liver function tests
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Urinalysis
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Urine drug screening
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Thyroid-stimulating hormone (TSH)
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Ceruloplasmin
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HIV
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Venereal Disease Research Laboratory test (VDRL)
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Vitamin D (25-OH), folate (Vit B9) and methylmalonic acid (B12)
Additional tests based on clinical picture
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Brain CT or MRI (preferable over CT) should be clinically guided with co-occurring neurological symptoms
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Lumbar puncture, testing for serum neuronal autoantibodies, namely anti-N-methyl-d-aspartic acid (NMDA).
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Electroencephalography (EEG)
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Rheumatologic workup, including lupus (ANA antibodies) and paraneoplasic disorders
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Karyotype/genetic tests
Differential diagnosis
A diagnosis of a first-episode psychosis was established, and from the brief history, it was suggested that there was a gradual onset of symptoms. The lack of neurological findings and the patient’s long-term substance use render substance-induced psychotic disorder as a likely hypothesis. However, considering the age group and chronological manifestations of positive symptoms, a possible diagnosis of schizophrenia requires a longitudinal assessment.
Treatment
The patient was admitted to the psychiatric ward and started a pharmacological treatment with daily paliperidone 6 mg per os (PO), lorazepam 1 mg PO and folic acid 5 mg PO as well as ensuring hydration, parenthetically initially and then orally, to mitigate THC withdrawals. After 5 days, paliperidone was increased to 9 mg PO daily due to the persistence of delusional paranoia.
Pharmacological treatment was combined with psychosocial interventions. Illness self-management training was included in the treatment plan, including psychoeducation, behavioural interventions to facilitate adherence and a relapse prevention plan. The patient received psychoeducation based on group cognitive behavioural therapy and motivational interviewing on addictions, and participated in inpatient activities that were available to all patients. These continued after discharge with a more structured and individual approach.
Additionally, the patient participated in occupational therapy activities such as mental exercises and training to improve cognitive performance and functional outcomes.
He received counselling on the risks of drug use, particularly cannabinoids, alcohol, stimulants and hallucinogens. The causal role of substance misuse in schizophrenia is still controversial but there is evidence that prior use of some drugs, particularly cannabis, is associated with an increased risk of developing schizophrenia later on. Cannabis use has also been shown to precipitate psychotic relapse in people with schizophrenia who had previously achieved remission and is associated with a worse outcome and reduced treatment efficacy.12
Family support and education about the illness was provided to mitigate disease burden and promote recovery.
Outcome and follow-up
In the hospital, the patient achieved full remission of delusions after 7 days with no identifiable delusions or changes in perception. His family also confirmed clinical improvement and at discharge; he was instructed to maintain daily paliperidone 9 mg PO and folic acid 5 mg PO.
One month later, at the first reassessment, the patient showed no signs of psychotic symptoms and reported maintaining abstinence from cannabinoids. He had sold his business and was in the process of starting a new job, a life change that he welcomed. The patient’s father corroborated the information. However, in the previous week, he was non-compliant with the medication because he thought it caused him muscle weakness. A reduction of the antipsychotic dosage was attempted (paliperidone 6 mg PO daily) but in the following visit, 1 month later, the patient had stopped the medication due to the aforementioned reason. A switch to another antipsychotic was proposed but the patient refused all options. Despite this, he agreed to attend a follow-up appointment. Thus, an advanced rescue plan was developed in collaboration with the family in case of relapse, with more frequent appointments and additional psychoeducation about the disease.
Discussion
Literature on the prevalence of DMS is scarce, despite the fact that is has been described in the context of numerous aetiological conditions for decades. It can often occur in schizophrenia, bipolar and schizoaffective disorders, but there is a lack of adequate scientific data characterising these manifestations. Overall prevalence shows a wide variation among studies, that ranges from 15%–49% of patients who suffered from psychosis, like schizophrenia, to 18%–40% of patients with organic mental syndromes, and 11% with affective disorders.13–15 When comparing the different DMS, the information available seems to indicate that CS is the most common subtype, with almost two-thirds of all assessed cases, followed by Fregoli’s with 7%, intermetamorphosis with 4% and remaining presentations often being combined.7 9 13 15 Suffering from DMS is comprehensively linked to suspicion and hostility, which in turn can lead to major life changes and paranoia, as presented in this case. These syndromes are not static as they can evolve into another type or present themselves as a complex, combined presentation; but, what makes the syndrome of subjective doubles particularly interesting is its rarity.9 In fact, we found no study that estimates prevalence of this subtype despite being relatively common in medical articles, and even featuring in classical literature such as ‘The Double’, by Fyodor Dostoyevsky, one of the most famous examples that presents clear and precise descriptions of psychotic symptoms.16 In 2017, a significant study associated DMS more frequently with schizophrenia but found that it also occurs in several other conditions, such as depressive disorders or dementias, which adds further evidence to the probable neurological structural etiopathogenesis and narrows the division between psychiatric and neurologic-systemic disorders. Some studies have found DMS prevalence rates of 16.6% in Lewy Bodies Dementia, 15.8% in Alzheimer’s disease and Parkinson Disease even before tremors, rigidity and bradykinesia. Additionally, several neurological conditions and various organic disorders, including cerebrovascular disease, subarachnoid haemorrhage, head injury, epilepsy, pituitary tumours have also been associated with DMS.7 8 11 Drug use is associated with an increased risk of psychotic disorders with some conflicting evidence regarding the effect of THC in dopaminergic modulation and the role of dopamine in DMS.17
Knowledge regarding DMS phenomena is becoming more extensive. However, when we consider subtypes, this knowledge is still regarded as incomplete and at a preliminary level. When discussing the syndrome of subjective doubles, some authors point to overactivity in the perirhinal cortex that accounts for a hyperfamiliarity and may explain this perception, while frontal lobe dysfunction is implicated in the inability to self-monitor/regulate or make familiarity assessments.9 18 19 These impairments alter the feeling of familiarity and thus the ability to integrate memories correctly, creating beliefs impermeable to evidence against them.5 The treatment of DMS poses a significant obstacle because of the lack of guidelines and the scarcity of scientific publications.4 Many reported patients experience spontaneous resolution of their DMS once the primary cause is addressed, but some may persist with a considerable functional impact.20 In spite of potential spontaneous resolution, in paranoid psychotic patients, the risk of violence is often increased in line with their false beliefs. Violence is usually directed at a specific person who is perceived as hostile or a persecutor. Therefore, DMS may be a risk factor for violent behaviour directed at the delusionally misidentified person. Although in this case we did not carry out a structured assessment of the risk of violence, there are many instruments to assess the risk of violence, including the Psychopathy Checklist-Revised, the Historical Clinical Risk Management-20 and the Violence Risk Appraisal Guide some of the most used scales.21 22
Patient’s perspective
“Now that I am calmer, I can reflect on what happened, although I do not like to remember or talk about it. When I fell sick, I felt like everything I had done in my life was wrong. I felt I had to sort things out so that I could start all over again. I know my family thought I was weird, and my behaviour was out of character but at the time I didn’t understand their concern and I even got angry because I thought I was fine and they told me I wasn’t. Looking back, I think I wanted to make too many changes in my life at once.
When I was hospitalised, I was unhappy with what my family had done to me and I thought they wanted to make me look crazy. However, when I was in hospital I managed to calm down, take a deep breath and realise that I had become detached from reality. Although the doctors explained to me that cannabis may have had an impact on my condition, I still think that what happened to me was due to overworking and the fact that I was living a very stressful life and deep down that was not what I wanted.”
Learning points
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The syndrome of subjective doubles is a rare example of delusional misidentification syndromes (DMS) which was historically associated with psychiatric disorders, but it is often present in other medical (non-psychiatric) conditions.
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Recent research suggests overactivity in the perirhinal cortex and frontal lobe dysfunction.
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When screening patients with DMS with a first-episode of psychosis, thorough medical examination should always be carried out.
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There are reports of spontaneous resolution of DMS, but in some patients it may persist and have a considerable functional impact.
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Very little has been published about treatment for DMS, so further investigation is needed to improve knowledge of these syndromes.
Ethics statements
Patient consent for publication
Footnotes
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Twitter @pedromorgado
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Contributors JLMQ, CMP and CPF contributed equally to this paper. PM, JLMQ and CMP were involved in the clinical diagnosis and patient's initial care. CPF is involved in the subsequent follow-up of the patient. JLMQ, CMP and CPF authors drafted the manuscript. PM substantially contributed to its revision as did JLMQ, CMP and CPF.
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Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
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Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
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Competing interests PM has received in the past 3 years grants, CME-related honoraria, or consulting fees from Angelini, AstraZeneca, Bial, Biogen, DGS-Portugal, FCT, FLAD, Janssen-Cilag, Gulbenkian Foundation, Lundbeck, Springer Healthcare, Tecnimede, Viatris and 2CA-Braga.
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Provenance and peer review Not commissioned; externally peer reviewed.
- © BMJ Publishing Group Limited 2022. No commercial re-use. See rights and permissions. Published by BMJ.
References
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