A Rare Case of Capgras Syndrome in Moyamoya Disease.

Moyamoya disease is a rare cerebrovascular disorder with unknown etiology and psychiatric symptoms occasionally manifest initially. Capgras syndrome is a unique neuropsychiatric symptom that is a delusional misidentification of a familiar person replaced by an identical imposter. We report the case of a 51-year-old woman with frontal lobe ischemic stroke caused by moyamoya disease, presenting with Capgras syndrome. Dysfunction of frontal areas may be attributable to development of Capgras syndrome.

A Case of Capgras Syndrome With Frontotemporal Dementia.

Capgras syndrome (CS), also called imposter syndrome, is a rare psychiatric condition that is characterized by the delusion that a family relative or close friend has been replaced by an identical imposter. Here, we describe a 69-year-old man with CS who presented to the Kemal Arikan Psychiatry Clinic with an ongoing belief that his wife had been replaced by an identical imposter. MRI showed selective anterior left temporal lobe atrophy. Quantitative EEG showed bilateral frontal and temporal slowing. Neuropsychological profiling identified a broad range of deficits in the areas of naming, executive function, and long-term memory. On the basis of these findings, we diagnosed frontotemporal dementia. This case demonstrates that CS can clinically accompany frontotemporal dementia.

Cognitive neuropsychiatric analysis of an additional large Capgras delusion case series.

INTRODUCTION: Although important to cognitive neuropsychiatry and theories of delusions, Capgras delusion has largely been reported in single case studies. Bell et al. [2017. Uncovering Capgras delusion using a large scale medical records database. British Journal of Psychiatry Open, 3(4), 179-185] previously deployed computational and clinical case identification on a large-scale medical records database to report a case series of 84 individuals with Capgras delusion. We replicated this approach on a new database from a different mental health service provider while additionally examining instances of violence, given previous claims that Capgras is a forensic risk. METHODS: We identified 34 additional cases of Capgras. Delusion phenomenology, clinical characteristics, and presence of lesions detected by neuroimaging were extracted. RESULTS: Although most cases involved misidentification of family members or partners, a notable minority (20.6%) included the misidentification of others. Capgras typically did not present as a monothematic delusion. Few cases had identifiable lesions with no evidence of right-hemisphere bias. There was no evidence of physical violence associated with Capgras. CONCLUSIONS: Findings closely replicate Bell et al. (2017). The majority of Capgras delusion phenomenology conforms to the "dual route" model although a significant minority of cases cannot be explained by this framework.

[Delusions: current psychodynamic and neurocognitive approaches]. / Wahn: aktuelle psychodynamische und neurokognitive Ansätze.

BACKGROUND: The symptom "delusions" is a central psychopathological symptom in psychiatric diseases. Since the beginning of psychiatry various disciplines have attempted to explain and understand delusions but even now no generally accepted definition of this phenomenon exists. AIM: A comprehensive review of current psychopathological and neurobiological theories of delusions is given. MATERIAL AND METHODS: PubMed and Google scholar searches were performed using the keywords "delusion", "psychodynamic" and "neurobiology", both in English and German. Relevant German textbooks of psychiatry were also included. DISCUSSION: A differentiated perspective of the phenomenon of delusions appears to be necessary to approach this complex and fascinating symptom. A one-dimensional approach does not do justice to the complexity of delusions. The various explanatory approaches can increasingly be linked to each other and are no longer considered to be mutually exclusive.

[Delusional misidentification syndromes: A factor associated with violence? Literature review of case reports]. / Les syndromes délirants d'identification : un facteur associé à la violence ? Revue de littérature à partir d'études de cas.

OBJECTIVES: Delusional misidentification syndromes (DMS) correspond to the delusional belief of misidentification of familiar persons, places or objects and to the conviction that they have been replaced or transformed. Several cases of patients who developed violent behavior while suffering from DMS have been published. This led some authors to consider patients with DMS at risk of violence. However, only a few studies have focused on the potential relationship between violence and DMS. The aim of our study was to explore this relationship with a literature review of published cases of patients having committed violent acts associated to DMS. METHODS: A systematic literature search was conducted on PubMed up to January 2017 using the following term combination "misidentification" and "violence" Fifteen cases of patients with DMS who had committed violent acts were identified. The data from these descriptions were analyzed and synthetized. RESULTS: Most of the patients were men with a diagnosis of schizophrenia and Capgras syndrome. Acts of violence were severe with a relatively high number of murders or attempted murders. For half of the patients these violent acts were perpetrated with weapons. Victims were regularly the patient's family members and the assaults were usually not planned. Delusional syndromes often progressed for several years. Importantly, substance abuse, which is known to increase the risk of violence in patients with schizophrenia, was only observed in two patients. CONCLUSION: DMS are associated with several risk factors of violence, such as a diagnosis of schizophrenia, specific delusions including megalomania, persecution, negative affects and identified targets. Despite this risk for severe violence, there are no existing guidelines on how to assess and treat DMS in schizophrenia. Accordingly, we propose (1) the establishment of formal diagnostic criteria, (2) the development of rigorous research on these syndromes and (3) the integration of DMS in assessment of violence risk in schizophrenic patients.

Late Onset First Episode Psychosis Emerging as Delusional Misidentification of Familiar Sacred Places During a Holy Pilgrimage: A Case Report and Literature Review.

BACKGROUND: The delusional misidentification syndromes (DMS) include a myriad of discrete but related syndromes, which have wide spectrum anomalies of familiarity. Several misidentification syndromes have been described in the psychiatric literature, the most common of these delusions are: the Capgras syndrome; the Fregoli syndrome; the syndrome of inter-metamorphosis; reduplicative paramnesia; and environmental reduplication. CASE PRESENTATION: The reported case highlights the emergence of late onset first episode psychosis in a Middle Eastern 65-year-old female who has no previous psychiatric history. The nature of psychosis was mainly delusions of misidentification and persecution. DISCUSSION: DMS are relatively rare and occur predominantly in association with schizophrenia and affective psychosis. Between 25 and 40% are associated with organic conditions such as dementia, head injuries, brain tumors, and epilepsy. Only three cases of misidentification of sacred places have been reported previously in the literature. This case report is the first to present a DMS, emerging as a late onset first episode psychosis during the sacred journey of Hajj. CLINICAL IMPLICATIONS: The reported case highlights the importance of early recognition and treatment of mental health conditions that may appear de novo during the Hajj sacred journey. Readily available psychiatric resources, psychotropic medications, and psycho-education may be pivotal in ensuring mental well-being of pilgrims, which is fundamental to maintain the mental capacity required for completing these journeys.

A case of Capgras syndrome with one's own reflected image in a mirror.

We report the case of a 78-year-old patient admitted to the hospital for behavioral and psychological disorders consisting in impressions of presence of a stranger located behind the bathroom mirror, who strikingly shared the patient's appearance but was considered a different person, yet. We discuss how this case can be interpreted as an atypical Capgras syndrome for his mirror image and how it suggests an adjustment of the classical dual-route model that sustains face recognition between covert (or affective) and overt neural pathways.

The doxastic shear pin: delusions as errors of learning and memory.

We reconsider delusions in terms of a "doxastic shear pin", a mechanism that errs so as to prevent the destruction of the machine (brain) and permit continued function (in an attenuated capacity). Delusions may disable flexible (but energetically expensive) inference. With each recall, delusions may be reinforced further and rendered resistant to contradiction. We aim to respond to deficit accounts of delusions - that delusions are only a problem without any benefit - by considering delusion formation and maintenance in terms of predictive coding. We posit that brains conform to a simple computational principle: to minimize prediction error (the mismatch between prior top-down expectation and current bottom-up input) across hierarchies of brain regions and psychological representation. Recent data suggest that delusions may form in the absence of constraining top-down expectations. Then, once formed, they become new priors that motivate other beliefs, perceptions, and actions by providing strong (sometimes overriding) top-down expectation. We argue that delusions form when the shear-pin breaks, permitting continued engagement with an overwhelming world, and ongoing function in the face of paralyzing difficulty. This crucial role should not be ignored when we treat delusions: we need to consider how a person will function in the world without them..

Capgras delusion with violent behavior in Alzheimer dementia: case analysis with literature review.

BACKGROUND: Capgras delusion (CD) has multiple etiologies including neurodegenerative disorders and can be associated with violent behavior. CD is a common complication of Alzheimer dementia (AD); however, CD with violent behavior is uncommon in AD. We report escalating violent behavior by a patient with advanced AD and CD who presented to the emergency department (ED) and required admission to an academic medical center. METHODS: Case analysis with PubMed literature review. RESULTS: A 75-year-old male with a 13-year history of progressive AD, asymptomatic bipolar disorder, chronic kidney disease, hypertension, hyperlipidemia, and benign prostatic hypertrophy presented to the ED with recurrent/escalating violence toward his wife, whom he considered an impostor. His psychotropic regimen included potentially inappropriate medications (PIMs) for geriatric/AD patients-topiramate/amitriptyline/chlordiazepoxide/olanzapine-that are associated with delirium, cognitive decline, dementia, and mortality. Renal dosing for topiramate, reduction in PIMs/anticholinergic burden, and substituting haloperidol for olanzapine resolved his violent behavior and CD. CONCLUSIONS: CD in AD is a risk factor for violent behavior. As the geriatric population in the United States grows, CD in patients with AD may present more frequently in the ED, requiring proper treatment. Pharmacovigilance is necessary to minimize PIMs in geriatric/AD patients. Clinicians and other caregivers require further education to appropriately address CD in AD.

Capgras' syndrome in first-episode psychotic disorders.

BACKGROUND: Misidentification phenomena, including the delusion of 'imposters' named after Joseph Capgras, occur in various major psychiatric and neurological disorders but have rarely been studied systematically in broad samples of modern patients. This study investigated the prevalence and correlated clinical factors of Capgras' phenomenon in a broad sample of patient-subjects with first-lifetime episodes of psychotic affective and nonaffective disorders. METHODS: We evaluated 517 initially hospitalized, first-episode psychotic-disorder patients for the prevalence of Capgras' phenomenon and its association with DSM-IV-TR diagnoses including schizophreniform, brief psychotic, unspecified psychotic, delusional, and schizoaffective disorders, schizophrenia, bipolar-I disorder and major depression with psychotic features, and with characteristics of interest including antecedent psychiatric and neurological morbidity, onset type and presenting psychopathological phenomena, using standard bivariate and multivariate statistical methods. RESULTS: Capgras' syndrome was identified in 73/517 (14.1%) patients (8.2-50% across diagnoses). Risk was greatest with acute or brief psychotic disorders (schizophreniform psychoses 50%, brief psychoses 34.8%, or unspecified psychoses 23.9%), intermediate in major depression (15%), schizophrenia (11.4%) and delusional disorder (11.1%), and lowest in bipolar-I (10.3%) and schizoaffective disorders (8.2%). Associated were somatosensory, olfactory and tactile hallucinations, Schneiderian (especially delusional perception), and cycloid features including polymorphous psychotic phenomena, rapidly shifting psychomotor and affective symptoms, pananxiety, ecstasy, overconcern with death, and perplexity or confusion, as well as rapid onset, but not sex, age, abuse history, dissociative features, or indications of neurological disorders. CONCLUSIONS: Capgras' syndrome was prevalent across a broad spectrum of first-episode psychotic disorders, most often in acute psychoses of rapid onset.

Importance of Capgras syndrome in shared psychotic disorder: a case report.

Shared psychotic disorder characterized by Capgras syndrome is an extremely rare condition. To our knowledge, there are only a few published papers on this condition. This paper presents a case of shared Capgras syndrome in two sisters. The inducer was a younger sister with schizophrenia, who passed on her Capgras delusion to her older sister after the death of their father. After committing a violent offense caused by Capgras delusion, a court ordered the sisters' involuntary admission to a psychiatric hospital. After being separated and receiving antipsychotic treatment, the sisters showed substantial improvement. However, shortly after hospital discharge, they stopped taking their medication and disappeared. After 15 years, their mother died and shortly afterwards, the sisters were re-admitted for forensic psychiatric evaluation after another violent crime caused by Capgras delusion. Timely recognition, adequate treatment and maintaining a therapeutic alliance could contribute to a better clinical course and outcome of this disorder, and reduce the risk of violent behavior.

Capgras syndrome in Dementia with Lewy Bodies.

BACKGROUND: Capgras syndrome is characterized by the recurrent, transient belief that a person has been replaced by an identical imposter. We reviewed clinical characteristics of Dementia with Lewy Bodies (DLB) patients with Capgras syndrome compared to those without Capgras. METHODS: We identified 55 consecutive DLB patients (11 cases with Capgras syndrome (DLB-C) and 44 cases without evidence of Capgras (DLB). Semi-structured interviews with the patient and an informant, neurological exams, and neuropsychological testing were performed. Caregivers were assessed for caregiver burden and depression. Primary comparisons were made between DLB-C and DLB. Exploratory analyses using stepwise logistic regression and bootstrap analyses were performed to determine clinical features associated with Capgras. RESULTS: DLB-C patients experienced more visual hallucinations and self-reported anxiety, had higher scores on the Neuropsychiatric Inventory, and were less likely to be treated with cholinesterase inhibitors at time of initial evaluation. Extrapyramidal symptoms and depression were not associated with Capgras. Caregivers of DLB-C patients had higher caregiver burden. DLB-C was associated with self-reported anxiety (OR = 10.9; 95% CI = 2.6-47.6). In a bootstrap analysis, clinical findings that were predictors of Capgras included visual hallucinations (log(OR) = 18.3; 95% CI = 17.9-19.3) and anxiety (log(OR) = 2.9; 95% CI = 0.31-20.2). CONCLUSIONS: Our study suggests that Capgras syndrome is common in DLB and usually occurs in the presence of anxiety and visual hallucinations, suggesting related etiopathogenesis. Early appreciation of Capgras syndrome may afford the opportunity to alleviate caregiver burden and improve patient and caregiver outcomes.

Capgras syndrome presenting in an adolescent girl in the Caribbean.

The case of a 16-year old Jamaican girl who presented to the psychiatric service of a general hospital with features of Capgras syndrome is presented. Her history, treatment, progress and relevant psychodynamic and neurocognitive issues are explored. This is the first known published case of an adolescent with Capgras syndrome from the Caribbean. The case highlights that the syndrome may occur in different cultural contexts and that clinicians should be sensitive to its existence in order to avert under-diagnosis or misdiagnosis.

Coexistence of different delusional misidentification syndromes in clinical practice: A case series.

OBJECTIVE: The objective of this study is to analyse the coexistence of several delusional misidentification syndromes in a clinical sample. METHODS: Over one year, a sample of six patients presenting two or more types of delusional misidentification syndromes was selected. All these patients were admitted to the psychiatric inpatient unit of a Spanish hospital. RESULTS: Despite the different diagnoses, the patients included presented different types of delusional misidentification syndromes, both hyperidentification and hypoidentification. Antipsychotic treatment was not very effective against these delusional misidentification syndromes. CONCLUSIONS: The coexistence of several delusional misidentification syndromes indicates that the aetiopathogenesis of the different types is similar. It is a field with important clinical implications, due to the poor response to treatment, as well as the possible medico-legal implications.

Delusion of inanimate doubles: description of a case of focal retrograde amnesia.

This paper reports the case of a patient, M.P., who developed delusion of inanimate doubles, without Capgras syndrome, after traumatic brain injury. His delusional symptoms were studied longitudinally and the cognitive impairments associated with delusion were investigated. Data suggest that M.P. did 'perceive' the actual differences between doubles and originals rather than 'confabulate' them. The cognitive profile, characterized by retrograde episodic amnesia, but neither object processing impairment nor confabulations, supports this hypothesis. The study examines the nature of object misidentification based on Ellis' and Staton's account and proposes a new account based on concurrent unbiased retrieval of semantic memory traces and biased recollection of episodic memory traces.

[The syndrome of Capgras: converging models]. / Het syndroom van Capgras: convergerende modellen.

BACKGROUND: Various theories have been proposed concerning the syndrome of Capgras. AIM: To search for a concept that is common to the various theories relating to the aetiology of the syndrome of Capgras. METHODS: We begin by describing a new case of Capgras syndrome that we encountered recently, and thereafter we searched the literature using PubMed and PsycINFO. RESULTS: Various explanatory models converge in that they all assign a central role to a changed affect. A sensation of 'alienation' at a conscious or subconscious level, together with inadequate evaluation of beliefs, seems plausible. All models demonstrate some form of 'splitting' i.e. the separate processing of information in time, anatomy or mental representation. CONCLUSION: Although there is no conclusive evidence to support any one of the hypotheses, the models display striking similarities. They complement each other.

[Recognizing delusional misidentification syndromes]. / Misidentificatiewanen herkennen.

Delusional misidentification syndrome is a less common neuropsychiatric symptom and can occur in different diseases as dementia and psychiatric diseases or as part of a somatic disease. It can be difficult to recognise and can give a high burden for the formal caregiver. In this article we describe three cases, the pathophysiology and the possible treatment of a delusional misidentification syndrome.

On abductive inference and delusional belief: why there is still a role for patient experience within explanations of Capgras delusion.

This paper aims to examine critically the explanatory model of delusional belief presented in Coltheart, Menzies, and Sutton's (2010) paper, "Abductive Inference and Delusional Belief". The authors acknowledge that certain aspects of the model are speculative. In return, I speculate over the likelihood that the model's emphasis on subpersonal processing adequately and coherently explains the symptoms (as best we know them) of patients with delusional misidentification (specifically, the Capgras delusion) and nondeluded equivalent patient groups. In addition, I offer an account of the Capgras delusion that is compatible with many of the tenets of Coltheart et al.'s model, but which preserves an important explanatory role for patient experience absent, and erroneously so, I contend, from the aforementioned model. The more integrated explanation I am proposing here also provides a number of pertinent empirical questions and testable hypotheses that could inform future models of delusional belief.

[Cenesthopathies: a disorder of background emotions at the crossroads of the cognitive sciences and phenomenology]. / Les cénesthopathies : un trouble des émotions d'arrière plan. Regards croisés des sciences cognitives et de la phénoménologie.

BACKGROUND: Cenesthesia and cenesthopathy have played a fundamental role in 19th and early 20th century French and German psychiatry. Cenesthesia refers to the internal, global, implicit and affective perception of one's own body. The concept of cenesthopathy was coined by Dupre and Camus in 1907 to describe a clinical entity characterized by abnormal and strange bodily sensations. AIMS: In this review, we examine the history of these concepts and the influence they have had on clinical, nosographical and phenomenological psychiatry and on cognitive neuropsychiatry. METHOD: We performed a narrative review of the published research literature. RESULTS: Classical French and German psychiatrists have written extensively on cenesthesia and cenesthopathy although these notions are no longer in the mainstream of contemporary psychiatry. However, they are still present in contemporary psychiatric nosography in the form of some controversial clinical entities clearly related to cenesthesia such as cenesthetic schizophrenia, hypochondriacal monothematic delusions, or the Capgras and the Cotard syndromes. These clinical entities are all associated with a state of depersonalization. We point out the similarities between Ey's description of the depersonalization syndrome, especially in psychosis, and the characteristics of cenesthesia. Philosophers like Sartre or Merleau-Ponty have developed the concept of cenesthesia, and in particular have added new concepts like "body schema". Similarly, phenomenological psychiatrists like Minkowski or Tellenbach have attempted to describe psychiatric disorders associated with cenesthesia and have also proposed new concepts (i.e. atmospheric sense) in order to understand them better. More recently, cognitive neuropsychiatry has tried to discover the mechanisms, which cause or contribute to the genesis of delusions. The majority of delusion theories developed in cognitive neuropsychiatry consider that the explanation of monothematic delusions involves one or two explanatory stages. The first stage corresponds to an abnormal experience (the experiential stage) while the second is related to abnormal reasoning (the inferential stage). This theoretical first stage has been considered to be the result of a highly unusual or bizarre perceptual experience. According to the authors, this experience refers to a phenomenon of depersonalization, a loss of cenesthesia, or a loss of a feeling of familiarity. For example, the neurocognitive models of the Capgras and Cotard syndromes have in common the belief that they are both based on various kinds of unusual experiences. These unusual experiences are thought to include affective or emotional experiences. Capgras' syndrome is possibly triggered by an abnormal affective experience in response to the sight of closed-person's face. Similarly, the Cotard syndrome may result from a general flattening of affective responses to external stimuli. The inferential stage can also differentiate between the two syndromes. Some empirical validation has already been obtained in Capgras' syndrome but not yet in Cotard's syndrome. CONCLUSION: This review illustrates that the historical descriptions of cenesthesia and cenesthopathy remain relevant in contemporary neurocognitive models and more generally suggests that the comprehension of quite complex phenomena like delusion requires a multidisciplinary approach.