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The spectrum of psychiatric manifestations in subacute sclerosing panencephalitis: A systematic review of published case reports and case series

Published online by Cambridge University Press:  29 January 2024

Ravindra K. Garg Open the ORCID record for Ravindra K. Garg [Opens in a new window] ,
Sujita K. Kar Open the ORCID record for Sujita K. Kar [Opens in a new window] ,
Hardeep S. Malhotra ,
Shweta Pandey ,
Amita Jain ,
Imran Rizvi ,
Ravi Uniyal  and
Neeraj Kumar
Ravindra K. Garg*
Affiliation:
Department of Neurology, King George’s Medical University, Lucknow, India
Sujita K. Kar
Affiliation:
Department of Psychiatry, King George’s Medical University, Lucknow, India
Hardeep S. Malhotra
Affiliation:
Department of Neurology, King George’s Medical University, Lucknow, India
Shweta Pandey
Affiliation:
Department of Neurology, King George’s Medical University, Lucknow, India
Amita Jain
Affiliation:
Department of Microbiology, King George’s Medical University, Lucknow, India
Imran Rizvi
Affiliation:
Department of Neurology, King George’s Medical University, Lucknow, India
Ravi Uniyal
Affiliation:
Department of Neurology, King George’s Medical University, Lucknow, India
Neeraj Kumar
Affiliation:
Department of Neurology, King George’s Medical University, Lucknow, India
*
Corresponding author: Ravindra K. Garg; Email: garg50@yahoo.com
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Abstract

Data related to psychiatric manifestations in subacute sclerosing panencephalitis (SSPE) is currently available only in the form of isolated case reports. In this systematic review, we evaluated the spectrum of psychiatric manifestations and their impact on the course and outcome of SSPE. Data were obtained from 4 databases (PubMed, Embase, Scopus, and Google Scholar), with the most recent search conducted on March 27, 2023. The PRISMA guidelines were followed, and the PROSPERO registration number for the protocol is CRD42023408227. SSPE was diagnosed using Dyken’s criteria. Extracted data were recorded in an Excel spreadsheet. To evaluate the quality of the data, the Joanna Briggs Institute Critical Appraisal tool was employed. Our search resulted in 30 published reports of 32 patients. The mean age was 17.9 years. Schizophrenia, catatonia, and poorly characterized psychotic illnesses were the 3 most common psychiatric presentations that were seen in 63% (20/32) of cases. Catatonia was seen in 4 patients. Affective disorders, mania, and depression were reported among 22% (7/32) cases. In approximately 81% (26/32) cases, the course of SSPE was acute fulminant. Treatment with antipsychotic drugs had poor or no response. Out of 17 patients, who received antipsychotic drugs, 6 patients noted severe extrapyramidal adverse effects. SSPE often masquerades as a psychiatric disorder. Unresponsive psychiatric symptoms, early extrapyramidal signs, and progressive encephalopathy indicate SSPE.

Keywords

CatatoniaExtrapyramidal disorderMyoclonusMeaslesschizophreniaencephalitis
Type
Review
Information
CNS Spectrums , Volume 29 , Issue 2 , April 2024 , pp. 87 - 95
Copyright
© The Author(s), 2024. Published by Cambridge University Press

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References

Garg, RK, Mahadevan, A, Malhotra, HS, Rizvi, I, Kumar, N, Uniyal, R. Subacute sclerosing panencephalitis. Rev Med Virol. 2019;29(5):e2058.10.1002/rmv.2058CrossRefGoogle ScholarPubMed
Nair, SS, Vysakha, KV, Menon, RN, Sundaram, S. Adult-onset subacute sclerosing panencephalitis. Pract Neurol. 2021:practneurol-2020-002880.10.1136/practneurol-2020-002880CrossRefGoogle ScholarPubMed
Garg, RK, Rizvi, I, Malhotra, HS, et al. The spectrum of neuropsychiatric manifestations in subacute sclerosing panencephalitis: a systematic review of published case reports and case series. PROSPERO 2023 CRD42023408227. https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42023408227. Assessed March 27, 2023.Google Scholar
Joanna Briggs Institute. 2017 critical appraisal checklist for case reports. https://jbi.global/sites/default/files/2019-05/JBI_Critical_Appraisal-Checklist_for_Case_Reports2017_0.pdf. Assessed April 1, 2023.Google Scholar
Dyken, PR. Subacute sclerosing panencephalitis. Current status. Neurol Clin. 1985;3(1):179–196.10.1016/S0733-8619(18)31062-4CrossRefGoogle ScholarPubMed
Koehler, K, Jakumeit, U. Subacute sclerosing panencephalitis presenting as Leonhard’s speech-prompt catatonia. Br J Psychiatry. 1976;129:29–31.10.1192/bjp.129.1.29CrossRefGoogle ScholarPubMed
Moodie, JW, Philcox, D, Naudé, WD, Lipper, S, Kipps, A. Atypical presentation of subacute sclerosing panencephalitis in 3 patients. S Afr Med J. 1980;58(24):968–971.Google ScholarPubMed
Caplan, R, Tanguay, PE, Szekely, AG. Subacute sclerosing panencephalitis presenting as childhood psychosis. J Am Acad Child Adolesc Psychiatry. 1987;26(3):440–443.10.1097/00004583-198705000-00029CrossRefGoogle ScholarPubMed
Salib, EA. Subacute sclerosing panencephalitis (SSPE) presenting at the age of 21 as a schizophrenia-like state with bizarre dysmorphophobic features. Br J Psychiatry. 1988;152:709–710.10.1192/bjp.152.5.709CrossRefGoogle ScholarPubMed
Duncalf, CM, Kent, JN, Harbord, M, Hicks, EP. Subacute sclerosing panencephalitis presenting as schizophreniform psychosis. Br J Psychiatry. 1989;155:557–559.10.1192/bjp.155.4.557CrossRefGoogle ScholarPubMed
Mattinson, PJ. Suspected SSPE - a delayed psychiatric presentation. Br J Psychiatry. 1989;154:116–118.10.1192/bjp.154.1.116CrossRefGoogle ScholarPubMed
Forrest, G, Stores, G. Subacute sclerosing panencephalitis presenting with psychosis and possible sexual abuse. Eur Child Adolesc Psychiatry. 1996;5(2):110–113.10.1007/BF01989503CrossRefGoogle ScholarPubMed
Jähnel, M. Paranoid-halluzinatorische Psychose als Erstmanifestation einer subakuten sklerosierenden Panenzephalitis (SSPE) bei einem 19-jährigen Mann. Psychiatrische Praxis. 2003;30(S 2):70–72.Google Scholar
Datta, SS, Jacob, R, Kumar, S, Jeyabalan, S. A case of subacute sclerosing panencephalitis presenting as depression. Acta Neuropsychiatr. 2006;18(1):55–57.CrossRefGoogle ScholarPubMed
Kayal, M, Varghese, ST, Balhara, YP. Psychiatric manifestation of SSPE. J Neuropsychiatry Clin Neurosci. 2006;18(4):560.10.1176/jnp.2006.18.4.560CrossRefGoogle ScholarPubMed
Theethira, TG, Ravat, SH, Shah, PU, Oak, PJ, Godge, YR, Chengappa, KR. Subacute sclerosing panencephalitis presenting as schizophreniform disorder. Acta Neuropsychiatrica. 2009;21(1):45–47.10.1111/j.1601-5215.2009.00357.xCrossRefGoogle Scholar
Baran, Z, Hanağasi, H, Uçok, A. An unusual late presentation of subacute sclerosing panencephalitis with psychotic symptoms. J Neuropsychiatry Clin Neurosci. 2010;22(1):123.E13.CrossRefGoogle ScholarPubMed
Aggarwal, A, Jain, M, Jiloha, R. Catatonia as the initial presenting feature of subacute sclerosing panencephalitis. J Neuropsychiatry Clin Neurosci. 2011;23(1):E29–E31.CrossRefGoogle ScholarPubMed
Aggarwal, A, Khandelwal, A, Jain, M, Jiloha, RC. Subacute sclerosing panencephalitis presenting as mania. Ann Indian Acad Neurol. 2011;14(2):120–121.10.4103/0972-2327.82801CrossRefGoogle ScholarPubMed
Altunkaynak, Y, Yıldırım, Z, Ertem, DH, Dirican, AC, Baybaş, S. Late onset subacute sclerosing panencephalitis: presenting psychosis. J Clin Anal Med. 2013;4(suppl2): 220–222.Google Scholar
Dayal, P, Balhara, YP. Catatonia as presenting clinical feature of subacute sclerosing panencephalitis. J Pediatr Neurosci. 2014;9(1):57–59.Google ScholarPubMed
Kartal, A, Kurt, AN, Gürkaş, E, Aydin, K, Serdaroğlu, A. Subacute sclerosing panencephalitis presenting as schizophrenia with an alpha coma pattern in a child. J Child Neurol. 2014;29(10):NP111–NP113.CrossRefGoogle ScholarPubMed
Erdogan, S, Yilmaz, R, Sorgun, MH, Aydin, N. Mania as a presenting clinical feature of subacute sclerosing panencephalitis. Acta Neurol Belg. 2015;115(4):803–805.CrossRefGoogle ScholarPubMed
Manoj, S, Mukherjee, A, Kumar, KV. Subacute sclerosing panencephalitis presenting with hypersexual behavior. Indian J Psychiatry. 2015;57(3):321–322.Google ScholarPubMed
Parmar, A, Ranjan, R, Sagar, R. Subacute sclerosing panencephalitis presenting with isolated positive psychotic and catatonic symptoms. Indian J Psychol Med. 2017;39(4):534–536.CrossRefGoogle ScholarPubMed
Tak, ZA, Celik, M, Kalenderoğlu, A, Sağlam, S, Ekmekci, B, Altun, Y. A case of sub-acute sclerosing panencephalitis presenting with nonspecific psychiatric symptoms. Turk Psikiyatri Dergisi. 2017;28(3):221.Google ScholarPubMed
Ahmad, J, Kar, SK, Uniyal, R. Acute and transient psychotic disorder as a rare early manifestation of late-onset sub-acute sclerosing panencephalitis. Neurol Psychiat Brain Res. 2018;30:35–38.CrossRefGoogle Scholar
Bhat, BA, Dar, SA, Hussain, A. Sub-acute sclerosing panencephalitis presenting as anti-depressant induced childhood mania. J Psychiatry. 2018;21:1000452.10.4172/2378-5756.1000452CrossRefGoogle Scholar
Reddy, B, Das, S, Guruprasad, S. Primary psychiatric manifestations of subacute sclerosing panencephalitis: a case report and literature review. Psychosomatics. 2018;59(4):408–412.CrossRefGoogle ScholarPubMed
Gokoglu, A, Gozdas, HT. Adult-onset subacute sclerosing panencephalitis presented with neuropsychiatric symptoms. J Coll Physicians Surg Pak. 2019;29(6):S29–S30.10.29271/jcpsp.2019.06.S29CrossRefGoogle ScholarPubMed
Arora, S, Jolly, AJ, Suhas, S, et al. Subacute sclerosing panencephalitis masquerading as schizophrenia: an example of diagnostic overshadowing in neuropsychiatry. Prim Care Companion CNS Disord. 2022;24(3):21cr02997.10.4088/PCC.21cr02997CrossRefGoogle ScholarPubMed
Sutar, R, Rai, NK. Revisiting periodic catatonia in a case of SSPE and response to intrathecal interferon: a case report. Asian J Psychiatr. 2020;51:101996.CrossRefGoogle Scholar
Chakraborty, K, Chatterjee, M, Bhattacharyya, R, Neogi, R. Case of subacute sclerosing pan-encephalitis (SSPE) presenting as catatonic schizophrenia. J Ind Assoc Child Adol Mental Health. 2021;17(2):204–209.Google Scholar
Kaur, G. Sub-acute sclerosing panencephalitis manifesting as psychiatric illness: a case report. Sch J Med Case Rep. 2021;10:938–941.CrossRefGoogle Scholar
Jain, P, Sehgal, A, Purohit, K, Sajan, C. SSPE presented as catatonia: case report. World J Pharma Res. 2022;11:1723–1726.Google Scholar
Espinola-Nadurille, M, Restrepo-Martínez, M, Bayliss, L, et al. Neuropsychiatric phenotypes of anti-NMDAR encephalitis: a prospective study. Psychol Med. 2022;10:1–9.Google Scholar
Hinotsu, K, Miyaji, C, Yada, Y, et al. The validity of atypical psychosis diagnostic criteria to detect anti-NMDA receptor encephalitis with psychiatric symptoms. Schizophr Res. 2022;248:292–299.CrossRefGoogle ScholarPubMed
Garg, RK, Kumar, N, Rizvi, I, Pandey, S, Malhotra, HS, Uniyal, R. Fulminant subacute sclerosing panencephalitis mimicking autoimmune encephalitis. Pediatr Infect Dis J. 2019;38(3):e64.10.1097/INF.0000000000002148CrossRefGoogle ScholarPubMed
Sachdev, P. Schizophrenia-like psychosis and epilepsy: the status of the association. Am J Psychiatry. 1998;155(3):325–336.10.1176/ajp.155.3.325CrossRefGoogle ScholarPubMed
Garakani, A. Commentary: psychiatric presentations of nonpsychiatric illness or treatment. J Psychiatr Pract. 2021;27(5):404–405.CrossRefGoogle ScholarPubMed
Ardila, A. Psychiatric disorders associated with acquired brain pathology. Appl Neuropsychol Adult. 2019;26(6):591–597.CrossRefGoogle ScholarPubMed
Dickerson, F, Stallings, C, Origoni, A, Copp, C, Khushalani, S, Yolken, R. Antibodies to measles in individuals with recent onset psychosis. Schizophr Res. 2010;119(1–3):89–94.10.1016/j.schres.2009.12.010CrossRefGoogle ScholarPubMed
Howes, OD, Cummings, C, Chapman, GE, Shatalina, E. Neuroimaging in schizophrenia: an overview of findings and their implications for synaptic changes. Neuropsychopharmacology. 2023;48(1):151–167.CrossRefGoogle ScholarPubMed
Insel, TR. Rethinking schizophrenia. Nature. 2010;468(7321):187–193.CrossRefGoogle ScholarPubMed
Tandra, HV, Roy, PS, Sharma, R, Bhatia, V, Saini, AG. Subacute sclerosing panencephalitis presenting as choreoathetosis and basal ganglia hyperintensities. Neurohospitalist. 2019;9(1):26–29.CrossRefGoogle ScholarPubMed
Lebon, S, Maeder, P, Maeder-Ingvar, M, et al. An initial MRI picture of limbic encephalitis in subacute sclerosing panencephalitis. Eur J Paediatr Neurol. 2011;15(6):544–546.CrossRefGoogle ScholarPubMed
Sawaishi, Y, Yano, T, Watanabe, Y, Takada, G. Migratory basal ganglia lesions in subacute sclerosing panencephalitis (SSPE): clinical implications of axonal spread. J Neurol Sci. 1999;168(2):137–140.10.1016/S0022-510X(99)00177-XCrossRefGoogle ScholarPubMed
Prashanth, LK, Taly, AB, Sinha, S, Ravi, V. Subacute sclerosing panencephalitis (SSPE): an insight into the diagnostic errors from a tertiary care university hospital. J Child Neurol. 2007;22(6):683–688.CrossRefGoogle ScholarPubMed
Sutton, T. Psychological testing: art, science and mysticism. PRECEDENT. 2010;99:39–43.Google Scholar
American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders: DSM-5. Washington, DC: American Psychiatric Association; 2013. doi:10.1176/appi.books.9780890425596.Google Scholar
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