Different SPECT findings before and after Capgras’ syndrome in interictal psychosis

doi:10.1016/j.yebeh.2006.04.016

Epilepsy & Behavior

Volume 9, Issue 1, August 2006, Pages 189-192

https://doi.org/10.1016/j.yebeh.2006.04.016 Get rights and content

Abstract

We herein report a case of Capgras’ syndrome observed in interictal psychosis in which the single-photon emission computed tomography (SPECT) findings before and after the appearance of the psychotic symptoms differed. SPECT with ^99mTc-hexamethyl propyleneamine oxine (HMPAO) revealed worsening of hypoperfusion in the entire right hemisphere after onset of the psychotic symptoms. The enhanced hypoperfusion demonstrated by SPECT in the present case seems to indicate a right interhemispheric disconnection resulting in the occurrence of Capgras’ syndrome.

Introduction

Capgras’ syndrome is one type of delusional misidentification syndrome (DMS), a category that includes Fregoli syndrome, the syndrome of intermetamorphosis, and the syndrome of subjective doubles [1], [2], [3], [4]. The delusional belief that familiar people and objects have been replaced by imposters is known as Capgras’ syndrome and was first described in 1923 by Capgras and Reboul-Lachaux. Cap-gras’ syndrome is relatively rare, occurring predominantly in the context of schizophrenia, and it was traditionally considered to have its origins in psychodynamic conflict. However, it has recently been estimated that between 25 and 40% of such cases are associated with organic disorders, including dementia, epilepsy, and cerebrovascular diseases. Neuroimaging evidence suggests a link between Capgras’ syndrome and right hemisphere abnormalities, particularly of the frontotemporal regions [1], [2], [3], [4]. From 2 to 7% people with epilepsy have been reported to suffer from psychosis, and the prevalence of psychosis in patients with temporal lobe epilepsy and/or refractory epilepsy varies from 10 to 19% in most studies [5]. We herein report a case of Cap-gras’ syndrome observed in interictal psychosis for which the single-photon emission computed tomography (SPECT) findings before and after the occurrence of psychotic symptoms differed. To the best of our knowledge, there have so far been no reports on SPECT findings in Capgras’ syndrome in interictal psychosis.

Section snippets

Case history

A 40-year-old, right-handed woman had her first episode of seizures at 7 years of age. Her natal, perinatal, and previous medical history was unremarkable. There was no past or family history of neuropsychiatric disorders. She experienced recurrent, complex partial seizures that evolved into generalized tonic–clonic seizures. At 9 years of age, she was referred to the pediatric neurology department for the first time. Her epileptic seizures were refractory and were occurring a few times per

Discussion

There have been some reports on DMS associated with epilepsy [6], [7], [8], [9]. Lewis described a case of interictal psychosis with Capgras’ syndrome, with bilateral occipitotemporal slow waves, more prominent in the right hemisphere [6]. Kim reported a case of postictal psychosis with Cap-gras’ syndrome, with right frontal slowing with intermittent epileptiform spikes [7]. Kanemoto reported a case of peri-ictal psychosis with Capgras’ syndrome, with clear-cut epileptiform discharges in the

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In fear of the most loved ones. A comprehensive review on Capgras misidentification phenomenon and case report involving attempted murder under Capgras syndrome in a relapse of a schizophrenia spectrum disorder
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While clinical examination is an integral part of the diagnostic procedure and brain Computed Tomography (CT) scan as well as Magnetic Resonance Imaging (MRI) are very important diagnostic tools in the field of neuroscience for the identification of organic conditions, there are a number of important areas where functional neuroimaging techniques, such as brain Positron Emission Tomography (PET) scan and Single Photon Emission Computed Tomography (SPECT) imaging, have the potential to provide relevant information to assist differential diagnosis and personalization of treatment to patients’ specific brain system pathophysiology.134–136 In four of the revised cases in the literature with Capgras symptomatology, functional imaging techniques disclosed important information on the functioning of the brain providing better insight to the pathophysiology of the underlying disorder and even more in different phases of the disease with concurrent alteration of the manifested Capgras symptomatology (Table 3).91,93,127,130 Physical violence is a common manifestation of mental disorders associated with Capgras syndrome, while anger and violence is most frequently directed towards the close family members, especially care-givers, on whom the delusional thoughts are predominantly focused (Table 1).
The present paper aims to describe a case of a schizophrenia spectrum disorder relapse leading to attempted murder, review literature, and investigate the epidemiological data and expression of violent behavior among Capgras-related incidents. 109 case reports deriving from various scientific areas dating from 1971 to 2017 were evaluated and juxtaposed with an equivalent comparison group of random psychiatry inpatients, who were examined for the same variables. Quantitative and qualitative differences were observed between samples. High level of interpersonal violence was disclosed among Capgras-related incidents, especially towards the main care-givers, with higher propensity among male patients. Homicidal behavior was also expressed in higher levels among male patients experiencing the Capgras delusion. A multidisciplinary approach is vital for the optimal management of these incidents. Further research on the pathophysiology of Capgras delusion with the utilization of functional imaging techniques is of exceptional significance for the understanding of issues of neuroscience.
The Anatomy of Delusion
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Delusions are maintained false beliefs despite compelling evidence to the contrary. They can occur in both psychiatric and neurologic disorders. They are distinct from hallucinations and confabulations and involve different neural circuitries and anatomy. Different content-specific delusions likely have different underlying anatomy and neurobiology; however, it is postulated that all delusions share a common anatomical and physiological denominator. This underlines the basis of the two-factor hypothesis, which proposes that delusions arise from two distinct brain dysfunctions. For example, formation of the Capgras delusion may require a dysfunction of both facial processing neural networks as well as hypothesis-appraisal networks. There may be some genetic factors influencing the occurrence and content of delusions.
Testing the connections within face processing circuitry in Capgras delusion with diffusion imaging tractography
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This means that the commissural connections of territories containing the main face processing regions were examined in the present study. This precludes abnormal interhemispheric connections as an explanation for CD in our patient, a hypothesis previously advanced for this disorder (Joseph, 1986; Horikawa et al., 2006) and for other delusional states (Filteau et al., 1991; Woodruff et al., 1995; Wang et al., 2008; Görgülü et al., 2010). On the other hand, the intact STS (not implicated by the gray matter VBM) and SLF (with a normal fiber tally) also discount a damaged ‘dorsal’ route for affective processing, at least as originally proposed (Bauer, 1984) which implicated a connection from temporal–parietal to frontal cortices.
Although Capgras delusion (CD) patients are capable of recognizing familiar faces, they present a delusional belief that some relatives have been replaced by impostors. CD has been explained as a selective disruption of a pathway processing affective values of familiar faces. To test the integrity of connections within face processing circuitry, diffusion tensor imaging was performed in a CD patient and 10 age-matched controls. Voxel-based morphometry indicated gray matter damage in right frontal areas. Tractography was used to examine two important tracts of the face processing circuitry: the inferior fronto-occipital fasciculus (IFOF) and the inferior longitudinal (ILF). The superior longitudinal fasciculus (SLF) and commissural tracts were also assessed. CD patient did not differ from controls in the commissural fibers, or the SLF. Right and left ILF, and right IFOF were also equivalent to those of controls. However, the left IFOF was significantly reduced respect to controls, also showing a significant dissociation with the ILF, which represents a selective impairment in the fiber-tract connecting occipital and frontal areas. This suggests a possible involvement of the IFOF in affective processing of faces in typical observers and in covert recognition in some cases with prosopagnosia.
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’” Delusional misidentification syndromes may be related to damage in the temporal, parietal, and frontal cortices, mostly in the right hemisphere [45,68], and have been shown to occur in patients with epilepsy postictally and interictally [69,70]. They may be interpreted as disturbances in the representation (or “cognitive map”) of other people, resulting in its disorganization, duplication, and incorrect location of self and others, similar to the time and place domains.
Orientation in time, space, and person is a fundamental cognitive faculty and the bedrock of neurological and psychiatric mental status examination. Nevertheless, research in orientation and disorientation is neglected in both cognitive science and neuropsychiatry. Specifically, it is still unclear whether disorientations in time, space, and person represent a failure of the same system or merely share a common nomenclature and whether these three domains of orientation depend on different psychological and neural systems. Here, we analyzed descriptions of patients with specific orientation failures associated with circumscribed cortical lesions, with a primary focus on epilepsy. The form of disorientation is analyzed according to its specific domain, the underlying neuropsychiatric disorder, and its anatomical correlate. Disorientations in the different domains are classified as self-referenced (incorrect self-localization) or nonself-referenced (incorrect localization or knowledge of other places, events, and people). Analysis of the cognitive and neural systems disturbed in these patients suggests that disorientation in one or several domains may be related to a failure in a specific brain mechanism localized mostly in the right hemisphere, partially overlapping with the default mode network (mostly the medial and lateral parietal, medial temporal, and lateral prefrontal cortices), which processes essential self-related cognitive faculties such as orientation.
Working memory dysfunction in delusional disorders: An fMRI investigation
2014, Journal of Psychiatric Research
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Also, in MRI studies of schizophrenia, the reduction in the volume of the left superior temporal gyrus was related with positive symptoms (Barta et al., 1990; Gaser et al., 2004; Nestor et al., 2007), thought disorders (McCarley et al., 1993; Shenton et al., 1992), and neuropsychological executive deficits (Nestor et al., 2007, 1993). Similarly, in a case report of DD, SPECT scans showed reduced perfusion in the right temporal region (Horikawa et al., 2006; Narumoto et al., 2006), whereas another case study showed the left temporoparietal region was mainly involved (Caliyurt et al., 2004; Hayashi et al., 2004; Wada et al., 1999). Our study showed increased activation in the bilateral temporal lobes.
Delusional disorder (DD) is a rare and understudied psychiatric disorder. There is limited number of studies concerning cognitive characteristics in DD. Using an established working memory paradigm with variable levels of memory load, we investigated alterations in functional magnetic resonance imaging (fMRI) of brain regions in patients with DD.
This case control study included 9 patients with DD and 9 healthy control subjects matched for age, sex, and education level. Diagnosis of DD was confirmed using the Structured Clinical Interview for DSM-IV Axis I. The severity of the symptoms was evaluated using the Positive and Negative Syndrome Scale. All patients were asked to perform 0-back and 2-back tasks during fMRI experiments. Functional imaging was performed using the 3.0 T Philips whole-body scanner using an 8-channel head coil.
Participants with DD had less neural activation of the left dorsolateral prefrontal cortex in fMRI scans obtained during performance tasks. On the other hand, neural activation of the left and right superior temporal gyrus, left middle and inferior temporal gyrus, right and left posterior cingulate gyrus, right amygdala, left and right fusiform gyrus was more prominent in patients with DD in comparison with the control group.
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While it is known that prosopagnosia is due to damage to right occipito-temporal brain regions, especially the occipital face area (Bouvier & Engel, 2006; Rossion et al., 2003) the dysfunctional brain circuits in Capgras delusion are largely unknown. There are several single case reports using mostly computer tomography (CT) or positron emission tomography (PET) in patients with Capgras delusion which suggest structural and metabolic anomalies in mostly right sided frontal, temporal or parietal brain regions (Horikawa et al., 2006; Lebert et al., 1994; Lewis, 1987; Luca et al., 2013; Paillere-Martinot, Dao-Castellana, Masure, Pillon, & Martinot, 1994). Note that however not all studies found right lateralized structural anomalies (Huang, Liu, & Yang, 1999).
Perception of familiar faces depends on a core system analysing visual appearance and an extended system dealing with inference of mental states and emotional responses. Damage to the core system impairs face perception as seen in prosopagnosia. In contrast, patients with Capgras delusion show intact face perception but believe that closely related persons are impostors. It has been suggested that two deficits are necessary for the delusion, an aberrant perceptual or affective experience that leads to a bizarre belief as well as an impaired ability to evaluate beliefs. Using functional magnetic resonance imaging, we compared neural activity to familiar and unfamiliar faces in a patient with Capgras delusion and an age matched control group. We provide evidence that Capgras delusion is related to dysfunctional activity in the extended face processing system. The patient, who developed the delusion for the partner after a large right prefrontal lesion sparing the ventromedial and medial orbitofrontal cortex, lacked neural activity to the partner's face in left posterior cingulate cortex and left posterior superior temporal sulcus. Further, we found impaired functional connectivity of the latter region with the left superior frontal gyrus and to a lesser extent with the right superior frontal sulcus/middle frontal gyrus. The findings of this case study suggest that the first factor in Capgras delusion may be reduced neural activity in the extended face processing system that deals with inference of mental states while the second factor may be due to a lesion in the right middle frontal gyrus.

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Case ReportDifferent SPECT findings before and after Capgras’ syndrome in interictal psychosis

Abstract

Introduction

Section snippets

Case history

Discussion

Psychosomatics

Psychiatry Res

Cortex

Epilepsy Behav

A review of the phenomenology and cognitive neuropsychological origins of the Capgras syndrome

Int J Geriatr Psychiatry

The delusional misidentification syndromes

Br J Psychiatry

Psychiatric, neurological and medical aspects of misidentification syndromes: a review of 260 cases

Psychol Med

Case Report
Different SPECT findings before and after Capgras’ syndrome in interictal psychosis