The Coexistence of Capgras, Fregoli and Cotard's Syndromes in an ...

Case Report / Olgu Sunumu 149The Coexistence of Capgras, Fregoli and Cotard’sSyndromes in an Adolescent CaseCapgras, Fregoli ve Cotard’s Sendromu Birlikteli¤i Olan Bir Ergen Olgufiermin YALIN, Fatma VAROL TAfi, Taner GÜVEN‹RDokuz Eylül Üniversitesi T›p Fakültesi, Çocuk Ruh Sa¤l›¤› ve Hastal›klar› Anabilim Dal›, ‹zmir, TurkeyABSTRACTA 14 year old adolescent girl who was diagnosed with Capgras, Fregoli andCotard's syndromes was described. Her clinical presentation contained thesigns of all three disorders of delusional misidentification syndrome.Coexistence of the signs of all three disorders is very rare in adolescents. Inaddition to rarity, lack of information and experience about the etiology andtreatment of these situations challenge the clinicians. (Archives ofNeuropsychiatry 2008; 45: 149-51)Key words: Delusional misidentification syndrome, adolescent, psychoticsymptomsÖZETBu olgu sunumunda Capgras, Fregoli ve Cotard’s sendromlar›n›n özelliklerini tafl›yan14 yafl›nda bir ergen k›z olgu tan›mlanmaktad›r. Bu olguda gözlenen sanr›lar›n,sanr›sal yanl›fl tan›ma sendromlar›n›n belirtilerinden oldu¤u ve her üçbozuklu¤a ait bulgular› da içerdi¤i belirlenmifltir. Ergen yafl grubunda üç bozuklu¤aait belirtiler nadir görülmektedir. Ayr›ca s›k rastlanmayan psikiyatrik durumlarolmas›, etiyolojisi ve tedavisi hakk›ndaki bilgi ve deneyimlerin eksikli¤iklinisyenler aç›s›ndan zorluk yaratmaktad›r. (Nöropsikiyatri Arflivi 2008; 45: 149-51)Anahtar kelimeler: Ergen, psikotik belirtiler, sanr›sal yanl›fl tan›mlama sendromuIntroductionDelusional misidentification syndrome includes a group ofdisorders that present delusions about oneself, others, placesand objects. The Capgras syndrome is the most common type.This syndrome was defined as a delusional psychiatricsyndrome by Capgras and Reboul-Lachaux in 1923 (1). Its mainfeature is the temporary, repetitive or permanent delusionalbelief that his/her parents, partner etc., have been replaced byidentical -looking ‘fake’ persons (2). In order to discriminatebetween the real and imposter, the patient uses small physicalappearance and behavior differences under the influence ofhis/her misperceptions (3).The Fregoli syndrome is another delusional misidentificationdisorder (1). In this disorder, the patients misidentify unfamiliarpeople and places as familiar ones. For example, they confuse ahospital room with their home and strangers with familiar people(4). The third disorder is the Cotard syndrome where the patientsdeny their own existence or a particular body part (5).No satisfactory explanation has been found for the etiology ofthese three psychopathological states. They have been relatedwith many neuro-psychiatric or organic and neurologicaldiseases, including dementia, head trauma, epilepsy,cerebrovascular disease, neurodegenerative disease, mostcommonly lewy body disease and multiple sclerosis (3). Althoughthey were initially reported as rare conditions, recent studieshave shown that they occur more frequently thanestimated (5-7).The coexistence of some of these syndromes has beenreported in the literature (1). However, there is only onereported case that presents the features of all three syndromes(8). In our case report, a 14 year old girl who presentedsymptoms of all the three syndromes will be described.Case reportMK is a 14 year old girl attending the 8th grade in primaryschool. Insomnia, food refusal, inablility to recognize her parentsand relatives, dullness, slow reactions, frequently apologizingwithout appropriate reasons and apathy were the initialcomplaints, and she was urgently hospitalized for differentialdiagnosis and treatment.Her family reported that complaints began 2 weeks beforeshe was hospitalized and her sleep decreased incrementally,Address for Correspondence/Yaz›flma Adresi: Dr. fiermin Yal›n, Dokuz Eylül Üniversitesi T›p Fakültesi, Çocuk Ruh Sa¤l›¤› ve Hastal›klar› Anabilim Dal›, ‹zmir, TurkeyE-posta: drserminyalin@hotmail.com Received/Gelifl tarihi: 30.05.2008 Accepted/Kabul tarihi: 25.10.2008© Nöropsikiyatri Arflivi Dergisi, Galenos Yay›nc›l›k taraf›ndan bas›lm›flt›r. Her hakk› sakl›d›r. / © Archives of Neuropsychiatry, Published by Galenos Publishing. All rights reserved.

150Yal›n et al.The Coexistence of Capgras, Fregoli and Cotard’s Syndromes in an Adolescent CaseArchives of Neuropsychiatry 2008; 45: 149-51Nöropsikiyatri Arflivi 2008; 45: 149-51she had not slept for the previous 2-3 days and staredmeaninglessly. She refused to eat, did not permit her parents toeat, had exaggerated fears, had thoughts that her parents andherself would be harmed, didn't recognize her parents in theprevious 2-3 days, wanted to go to her ‘real’ parents, thought thatstrangers had replaced her parents and would not cooperate.She had not had similar complaints in the past, but during theprevious 3 months she had been worrying about her academicperformance in the high school entrance exam. There were noproblems reported in her family and peer relations, she was alsodoing well academically at school.In the initial evaluation for the mental status exam, she had afearful face and was not communicating verbally. Her affect wasdull and her mood was reported as very anxious. Her attentionperiod was very short and her orientation, judgment and realitytesting were impaired. She was suffering from paranoidpersecutiondelusions and she also had a derealization anddepersonalization thought context. Additionally, her thoughtprocess and psychomotor activities were retarded.In order to differentiate the diagnosis, biochemical andendocrinological tests, electroencephalography (EEG),computerized brain tomography (CBT), magnetic resonanceimaging of brain (MRI) and pediatric neurology consultationswere carried out. There were no abnormalities reported in theneurological examination or laboratory tests. During thepatient’s stay in our unit, the most remarkable sign was herinability to recognize her parents and her delusional thoughtsthat they were not her real parents. When she was with herparents, she thought that they were doubles or fakes who hadreplaced her real parents. However, when she was on thephone with them, she did not express any suspicions aboutthem. She had delusional thoughts with a persecutory context,such as the hospital was not a real one, the medical team werenot hospital staff, everything was a play in which she wouldeventually be harmed. She also had reference delusions thatpeople were talking about and laughing at her. In addition, shehad more delusional thoughts such as that she knew some ofthe patients in the unit and members of the team, and thatactually she was dead and her soul was wandering aroundwaiting to be punished. Due to her mother’s pregnancy thepatient was over-anxious about her health.Having excluded the organic etiology, risperidonetreatment with 2 mg per day was begun and the daily dosegradually increased to 6mg/day. As there was no positivetreatment response to 6mg/day risperidone after 3 weeks, thiswas replaced with another atypical antipsychotic, olanzapine.Despite the fact that psychotic symptoms diminished, a clinicalmanic shift appeared. Valproate was added to the treatmentwhen the manic state of the patient gradually becamemanifest after commencing olanzapine. Following the additionof valproate, olanzapine was decreased while quetiapinetreatment was started and the quetiapine dose was graduallyincreased to 1500 mg per day. On discontinuing olanzapine, themanic symptoms disappeared. In the process, the patient’spsychotic symptoms recovered significantly and eventually shewas discharged without any symptoms.DiscussionDuring the treatment period in the unit, it was observed thatour patient had symptoms of Capgras, Cotard and FregoliSyndromes. The main clinical characteristics of the Capgrassyndrome is a delusional belief that familiar persons arereplaced by strangers (2).The patient refers to some small,misinterpreted physical and behavioral differences in her mindto differentiate the familiar persons from strangers (3). Whenpatients are questioned about their delusional ideas, they findthem unbelievable and weird, but these delusional thoughtscontinue (2). The most dominant delusional thought in our casewas that strangers had taken the place of her parents. Shemaintained that her visitors (real parents) looked the same as herparents, but their behavior and way of looking were different andshe insisted on seeing her real parents.The Cotard syndrome is characterized by delusionalthoughts of denying one’s own existence or particular parts ofhis/her body (5, 9). In the literature, 89% of the cases haddepressive symptoms, 69% had nihilistic delusions about theirexistence, 65% had anxiety symptoms, and 63% had feelings ofguilt (5). Our patient said that she had died and didn’t exist.Meanwhile, she thought this was all her fault and kept apologizing.She was suffering from significant anxiety regarding herdelusional experiences and her mother’s state of health.In the Fregoli syndrome, patients have delusional beliefs thatthey know quite unfamiliar persons and places very well. Theycan perceive the hospital room as their home, or a stranger astheir relative (4). Our case believed that some patients in the unitwere her parents and some of the staff were her relatives.Especially in the Capgras Syndrome, there are someadditional symptoms such as seen in our case. Paranoid ideas,derealization and depersonalization are the frequentdisturbances (1, 7). One study found 83% paranoid delusions and43% derealization and depersonalization in Capgras syndromecases (7).Although initial studies found that these syndromes wererare, subsequent investigations showed that they were morefrequent. A study conducted in an acute psychiatric servicefound that the Capgras syndrome rate was 2.5% and in anotherstudy which evaluated the previous 5 years of patients’ recordsin a university hospital, the rate was 1.3% (6, 7). This syndromecan affect both genders at all ages (6). However, some authorsfound that it was more frequent in women (2).Berios et al reviewed the literature since 1955 and reported100 Cotard syndrome cases. The average age of these caseswas 52 years and the youngest patient was 16 years (5). Asreported in the literature, the Cotard syndrome had a differentclinical course in young cases. Its frequency and relation withother disorders were not definite. It was reported that thesyndrome was more frequent in girls and the girl/boy ratio was12/7 (9). Mojtabei (1994) reviewed the literature in 1994 andreported 34 Fregoli syndrome cases. Their average age was 34.9years and mainly male (4).There has been no definite explanation about the etiology ofall these psychopathological conditions. In the literature, theinitial studies regarding the Capgras syndrome attributed the

Archives of Neuropsychiatry 2008; 45: 149-51Nöropsikiyatri Arflivi 2008; 45: 149-51Yal›n et al.The Coexistence of Capgras, Fregoli and Cotard’s Syndromes in an Adolescent Case 151causation psychoanalytically but subsequent authorsemphasized diffuse and localized brain lesions, whereas someauthors reported the association of two different causations (10).In the literature 25-40% of the Capgras syndrome cases had anorganic etiology. Brain imaging studies are essential in order toclarify the organic etiology. The lesion is more frequent in thefrontal, parietal and temporal lobes (2). In etiological studies ofthe Cotard syndrome, the disorder was found to be related withorganic mental disorders such as psychotic major depression,schizophrenia, epilepsy, encephalitis and pre-senility (9).Schizophrenia was the most frequent cause in the FregoliSyndrome. Psychosis with an organic etiology, affectivedisorders and other psychotic disorders were also frequentco-morbidities (4). In our case, all investigations to determine anorganic etiology were undertaken, but no pathology was found.Although the neurological examination was normal, the EEGshowed non-specific abnormalities. These abnormal signs couldbe insufficient to explain the symptoms of the patient but mightbecome very important during the follow-up. In addition, it wasemphasized that there was a relation between organic causesand the age of the patient, and the age could be important fordetermining the etiology in the Capgras syndrome. An organicetiology was found in 4 of 13 Capgras cases in a study with anaverage patient age of 33 years, and in another study themajority of the Capgras patients with an average age of 56 yearshad an organic pathology. In view of the literature regarding theonset age of the psychopathology, the probability of organicetiology in our case is very low (7).Psychodynamic explanations of the origins of the Capgrassyndrome consist of four basic groups; unresolved oedipalproblems in women, alienation and other affective problems,problems related with ambivalence and pathological separationof internal object representatives (10). Our patient's historyincludes the anxiety of preparation for high school exams andambivalent feelings about her mother’s pregnancy in theprevious year. During such a difficult time in her life, theadolescent might feel alienated and lonely when her ambivalentanxiety and oedipal conflicts have been stirred up by currentchallenges. The patient could handle the intense feelings onlyby denying and projecting as a split part to, her parents. As aresult, she might conclude in a delusional way that the mostconvincing explanation of this situation was they were not herreal parents.An overview of the clinical course of these disorders showsthat it could be either temporary or permanent. In the literature,the treatment models regarding these disorders are limited tocase reports. There were positive results with ECT in 9 caseswith the Cotard syndrome (9). Literature related with thetreatment of the delusional misidentification syndromesindicates that they are responsive to the atypical antipsychoticssuch as olanzapine, sulpiride and others (1, 3). There are positivetreatment outcome reports with 8 mg per day sulpiride in onecase and 5 mg per day olanzapine with another (11, 12). If anorganic cause has been found, it is advisable to treat thisinitially. Our case had an acute start and signs were resistant topharmacological treatment. Risperidone was ineffective andsymptoms were diminished with olanzapine and quetiapine.With olanzapine treatment, delusional misidentificationsyndrome symptoms were cleared but in the meantime manicsymptoms appeared. Despite the additional mood stabilizeragent added to her treatment plan there was no clinicalresponse, so eventually olanzapine treatment was changed toquetiapine. Psychotic symptoms reappeared with thetermination of olanzapine. However, by regulating the quetiapinedaily dosage, the patient became free of delusionalmisidentification syndrome symptoms.In this report, the case was discussed in terms of thecoexistence of the characteristics of all three syndromes. It isbelieved that most frequently there may be an organic causeunderlying the clinical presentation, but in our case there was noevidence for this. Such cases are a challenge for the cliniciansdue to the lack of information and experience regarding theetiology and treatment. It is essential to conduct both descriptiveand long-term follow-up studies in order to enrich existingknowledge and experiences in the etiology and treatment ofthese cases.References1. Feinberg TE, Roane DM. Delusional misidentification. Psychiatr Clin NAm 2005; 28: 665-83.2. Edelstyn NMJ, Oyebode FA. review of the phenomenology andcognitive neuropsychological origins of the Capgras syndrome. Int JGeriat Psychiatry 1999; 14: 48-59.3. Bourget D, Whitehurst L. Capgras syndrome: a review of theneurophysiological correlates and presenting clinical features incases involving physical violence. Can J Psychiatry 2004; 49: 719-25.4. Mojtabei R. Fregoli syndrome. Aust N Z J Psychiatry 1994; 28: 458-62.5. Berrios GE, Luque R. Cotard’s syndrome: analysis of 100 cases. ActaPsychiatr Scand 1995; 91: 185-8.6. Huang TL, Liu CY, Yang YY. Capgras syndrome: analysis of nine cases.Psychiatry and Clinical Neurosciences 1999; 53: 445-60.7. Tamam L, Karatas G, Zeren T ve ark. The prevalence of Capgrassyndrome in a university hospital setting. Acta Neuropsychiatrica2003a; 15: 290-5.8. Wolff G, McKenzie K. Capgras, Fregoli and Cotard’s syndromes andKoro in folie a deux. Br J Psychiatry 1994; 165: 842.9. Soultanian C, Perisse D, Révah-Levy A ve ark. Cotard’s syndrome inadolescents and young adults: a possible onset of Bipolar disorderrequiring a mood stabilizer? J Child Adolesc Psychopharmacol 2005;15: 706-11.10. Tamam L, Tamam Y, Ozpoyroz N. Capgras Sendromu: Bir olgu sunumuYeni Symposium 2003b; 41: 51-3.11. Tueth MJ, Cheong JA. Successful treatment with pimozide of Capgras’Syndrome in an elderly male. J Geriatr Psychiatry Neurol 1992; 5: 217-9.12. Butler PV. Diurnal variation in Cotard's Syndrome (copresent withCapgras delusion) following traumatic brain injury. Aust N Z JPsychiatry 2000; 34: 684-7.