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LETTERS TO THE EDITOR
Year : 2021  |  Volume : 24  |  Issue : 4  |  Page : 600-601
 

Pediatric acute-onset neuropsychiatric syndrome with capgras syndrome


1 Department of Pediatrics, Pediatric Neurology Division, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India
2 Department of Psychiatry, All India Institute of Medical Sciences, Rishikesh, Uttarakhand, India

Date of Submission04-Sep-2020
Date of Acceptance11-Oct-2020
Date of Web Publication04-Feb-2021

Correspondence Address:
Dr. Prateek Kumar Panda
Department of Pediatrics, Pediatric Neurology Unit, All India Institute of Medical Sciences, Rishikesh - 249203, Uttarakhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/aian.AIAN_959_20

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How to cite this article:
Sharawat IK, Panda PK, Gupta R. Pediatric acute-onset neuropsychiatric syndrome with capgras syndrome. Ann Indian Acad Neurol 2021;24:600-1

How to cite this URL:
Sharawat IK, Panda PK, Gupta R. Pediatric acute-onset neuropsychiatric syndrome with capgras syndrome. Ann Indian Acad Neurol [serial online] 2021 [cited 2021 Oct 25];24:600-1. Available from: https://www.annalsofian.org/text.asp?2021/24/4/600/308702




A nine-year-old previously healthy girl presented with acute onset behavioral changes in the form of obsessive preoccupation with the theme of contamination and compulsions of cleaning. She belonged to a family with middle socio-economic status, had one healthy younger male sibling, resisting in a nuclear family, with no significant family conflict, substance abuse, or financial issues, was studying in the fourth standard with average scholastic performance. There was no history of significant psychiatric illness in family members.

She also developed anorexia and emotional lability, in the form of easily getting annoyed when confronted about her actions and used to start crying suddenly in inappropriate situations. She started reaming more aloof and lost interest in study as well as other daily chores. Five days into the illness, there was an increase in abnormal behavior, and refused to recognize her parents. On further questioning, she told that her parents were replaced by some imposters and they are trying to hurt her. She had no other delusion, hallucination, other features of schizophrenia, but had mild symptoms of anxiety and depression according to the Childhood depression rating scale and screen for child anxiety disorders. She however used to answer queries by clinician appropriately. She had acute pharyngotonsillitis three weeks back, which was treated with a course of oral amoxicillin-clavulanic acid combination, for seven days by a private practitioner. Physical and neurological examination, serum electrolytes, endocrinal workup, magnetic resonance imaging (MRI) of the brain, sleep electroencephalogram, neurometabolic investigations, cerebrospinal fluid (CSF) examination including CSF autoimmune antibody profile and urine toxicology screen were noncontributory. Serum ASO titer was positive in high titers (768 IU), but serum C-reactive protein, ESR, cardiac and rheumatological examination including echocardiogram was normal. PANS (Pediatric Acute-onset Neuropsychiatric Syndrome) rating scale score was 102, suggestive of moderate severity. She was given psychological support and her psychopathology was closely monitored, with a probable diagnosis of the Pediatric acute-onset neuropsychiatric syndrome with Capgras syndrome (Organic delusional [schizophrenia-like] disorder” F06.2, according to ICD code). Over the next four weeks, the obsessive-compulsive features reduced in severity, her appetite improved, and she restarted acknowledging her parents.

Capgras syndrome is characterized by a specific monothematic delusion in which the patient believes that his/her relatives have been substituted by impostors who bear a close resemblance with the originals.[1] A systematic review of 255 published adult cases showed that it can occur with schizophrenia, schizoaffective disorder, brain damage due to various lesions like tumor, trauma, especially to the frontal/temporal lobe, and in neurodegenerative diseases like Alzheimer's disease.[2] It is more common in females like our case, but extremely rare in children, and in published literature, we could detect only six publications in children. None of these cases were formally evaluated for autoimmune encephalitis and PANS. These cases had been treated successfully with neuroleptic/atypical antipsychotics, SSRIs, and psychotherapy previously. Only 6 out of 107 patients in the review by Berson et al. were adolescents, while Kourany et al. also described a 16-year-old adolescent girl with dysmorphic delusions and Capgras syndrome.[3] An 8½-year-old boy has been described in literature who believed he was a double of himself.[1] Mazzone et al. have described a favorable response to risperidone and SSRI in an 11-year-old girl.[4] Almost all these patients had other delusions, along with emotional and behavioral problems, but none had obsessive-compulsive features or excessively decreased oral intake required essentially to suggest the diagnosis of PANS.[5] However, like our case, all pediatric cases had acute/subacute onset and none had definitive features of schizophrenia persistently.

Children with PANS also may have tics, emotional lability and/or depression, anxiety, irritability, aggression and/or severely oppositional behaviors, behavioral (developmental) regression, deterioration in school performance, and somatic signs and symptoms, including sleep disturbances, enuresis, or urinary frequency.[6] Children with PANS often have an unpredictable or relapsing-remitting course and if not treated timely than may lead to permanent debilitation. Although the majority of cases are triggered by infections, psychological, autoimmune, endocrine, neurometabolic, and even some other unknown non-infectious triggers have been implicated, but essentially this is a diagnosis of exclusion[6],[7] Mild cases can be treated with antipsychotics successfully, while severe cases require immunomodulation including corticosteroids. We could not find any case of Capgras syndrome in a child with PANS in published literature, although Schlansky has reported catatonia in a case of PANS.[8] Clinicians need to be aware of this rare monothematic delusion, as complete resolution can be achieved with timely treatment.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Kourany RFC. Capgras syndrome variant in an 8½-year-old boy. J Am Acad Child Psychiatry 1983;22:571-2.  Back to cited text no. 1
    
2.
Pandis C, Agrawal N, Poole N. Capgras' delusion: A systematic review of 255 published cases. Psychopathology 2019;52:161-73.  Back to cited text no. 2
    
3.
Berson RJ. Capgras' syndrome. Am J Psychiatry 1983;140:969-78.  Back to cited text no. 3
    
4.
Mazzone L, Armando M, De Crescenzo F, Demaria F, Valeri G, Vicari S. Clinical picture and treatment implication in a child with Capgras syndrome: A case report. J Med Case Reports 2012;6:406.  Back to cited text no. 4
    
5.
Jackson RS, Naylor MW, Shain BN, King CA. Case study capgras syndrome in adolescence. J Am Acad Child Adolesc Psychiatry 1992;31:977-83.  Back to cited text no. 5
    
6.
Wilbur C, Bitnun A, Kronenberg S, Laxer RM, Levy DM, Logan WJ, et al. PANDAS/PANS in childhood: Controversies and evidence. Paediatr Child Health 2019;24:85-91.  Back to cited text no. 6
    
7.
Josephs KA. Capgras syndrome and its relationship to neurodegenerative disease. Arch Neurol 2007;64:1762-6.  Back to cited text no. 7
    
8.
Schlansky K, Facer B, Tanguturi YC, Cundiff AW, Fuchs DC. Pediatric acute-onset neuropsychiatric syndrome and catatonia: A case report. Psychosomatics 2020;61:86-91.  Back to cited text no. 8
    




 

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