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Journal of Pediatric Neurology 2022; 20(03): 182-187
DOI: 10.1055/s-0041-1728774
Original Article

Resurgence of Subacute Sclerosing Panencephalitis: Case Series and Global Epidemiological Trends

Kavita Srivastava
1   Pediatric Neurology Unit, Department of Pediatrics, Bharati Vidyapeeth Deemed University Medical College, Pune, Maharashtra, India
,
Ekta Agarwal 
1   Pediatric Neurology Unit, Department of Pediatrics, Bharati Vidyapeeth Deemed University Medical College, Pune, Maharashtra, India
,
Surekha Rajadhyaksha
1   Pediatric Neurology Unit, Department of Pediatrics, Bharati Vidyapeeth Deemed University Medical College, Pune, Maharashtra, India
› Author Affiliations Funding This study was funded by Bharati Medical Foundation, Pune, India.
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Abstract

Resurgence of subacute sclerosing panencephalitis (SSPE)—case series and global epidemiological trends. We noted a recent increase in cases of SSPE admitted in our institute, even though they had received measles vaccination. We did a detailed study of our cases and compared with global epidemiological trends of SSPE in preimmunization and era of developed immunity. Out of total 23 cases of SSPE, 12 presented in the year 2017 alone, reflecting a steep rise in incidence. Sixteen patients had received measles vaccine and never had prior measles infection. Mean age of onset was 8.2 years and average time of progression to advanced stage of disease was 65 days. Global data showed similar trends, that is, earlier age of onset with a faster rate of progression in the postvaccination era as compared with prevaccine era. Possible mechanisms to account for this trend include an early wild measles infection in the critical age of 6 to 9 months, before vaccination. There is a changing epidemiological trend of SSPE in terms of lower age of onset and faster rate of progression, also reflected in global data. There is a need for multicenter studies to verify the findings and explore possible measures like lowering the age of measles vaccination to halt this alarming trend.

Keywords

subacute sclerosing panencephalitis - epidemiology - postvaccination era - measles


Publication History

Received: 19 December 2020

Accepted: 18 March 2021

Article published online:
01 June 2021

© 2021. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Garg RK, Karak B, Sharma AM. Subacute sclerosing panencephalitis. Indian Pediatr 1998; 35 (04) 337-344
    PubMedGoogle Scholar
  • 2 Kühne Simmonds M, Brown DW, Jin L. Measles viral load may reflect SSPE disease progression. Virol J 2006; 3: 49
    CrossrefPubMedGoogle Scholar
  • 3 Sardana V, Sharma D, Agrawal S. Subacute sclerosing panencephalitis revisited. Internat. J. of Basic and Applied Medical Sciences 2013; 3 (01) 225-241
    PubMedGoogle Scholar
  • 4 Malik B, Sharma FJ, Bhardwaj AK, Sharma A. Subacute sclerosing pan encephalitis despite adequate vaccination. Australas Med J 2012; 5 (07) 359-361
    CrossrefPubMedGoogle Scholar
  • 5 Saha V, John TJ, Mukundan P. et al. High incidence of subacute sclerosing panencephalitis in south India. Epidemiol Infect 1990; 104 (01) 151-156
    CrossrefPubMedGoogle Scholar
  • 6 Manayani DJ, Abraham M, Gnanamuthu C, Solomon T, Alexander M, Sridharan G. SSPE - the continuing challenge: a study based on serological evidence from a tertiary care centre in India. Indian J Med Microbiol 2002; 20 (01) 16-18
    PubMedGoogle Scholar
  • 7 Vaidya SR. Commitment of measles elimination by 2020: challenges in India. Indian Pediatr 2015; 52 (02) 103-106
    CrossrefPubMedGoogle Scholar
  • 8 Khetsuriani N, Sanadze K, Abuladze M, Tatishvili N. High risk of subacute sclerosing panencephalitis following measles outbreaks in Georgia. Clin Microbiol Infect 2020; 26 (06) 737-742
    CrossrefPubMedGoogle Scholar
  • 9 Dyken PR. Clinical expressivity in resurging SSPE: changing age of onset and new early symptoms. J Pediatr Neurol 2004; 2 (02) 53-56
    PubMedGoogle Scholar
  • 10 Dyken PR. Subacute sclerosing panencephalitis. Current status. Neurol Clin 1985; 3 (01) 179-196
    CrossrefPubMedGoogle Scholar
  • 11 Anlar B, Köse G, Gürer Y, Altunbaşak S, Haspolat S, Okan M. Changing epidemiological features of subacute sclerosing panencephalitis. Infection 2001; 29 (04) 192-195
    CrossrefPubMedGoogle Scholar
  • 12 Guler S, Kucukkoc M, Iscan A. Prognosis and demographic characteristics of SSPE patients in Istanbul, Turkey. Brain Dev 2015; 37 (06) 612-617
    CrossrefPubMedGoogle Scholar
  • 13 Okuno Y, Nakao T, Ishida N. et al. An epidemiological study of subacute sclerosing panencephalitis in Japan, 1976. Biken J 1978; 21 (A): 9-14
    PubMedGoogle Scholar
  • 14 Campbell H, Andrews N, Brown KE, Miller E. Review of the effect of measles vaccination on the epidemiology of SSPE. Int J Epidemiol 2007; 36 (06) 1334-1348
    CrossrefPubMedGoogle Scholar
  • 15 Abe Y, Hashimoto K, Iinuma K. et al. Survey of subacute sclerosing panencephalitis in Japan. J Child Neurol 2012; 27 (12) 1529-1533
    CrossrefPubMedGoogle Scholar
  • 16 Manning L, Laman M, Edoni H. et al. Subacute sclerosing panencephalitis in Papua New Guinean children: the cost of continuing inadequate measles vaccine coverage. PLoS Negl Trop Dis 2011; 5 (01) e932
    CrossrefPubMedGoogle Scholar
  • 17 Bojinova V, Dimova P, Belopitova L. et al. Subacute sclerosing panencephalitis in Bulgaria (1978-2002). Neuroepidemiology 2004; 23 (05) 254-257
    CrossrefPubMedGoogle Scholar
  • 18 Schönberger K, Ludwig MS, Wildner M, Weissbrich B. Epidemiology of subacute sclerosing panencephalitis (SSPE) in Germany from 2003 to 2009: a risk estimation. PLoS One 2013; 8 (07) e68909
    CrossrefPubMedGoogle Scholar
  • 19 Naruszewicz-Lesiuk D, Wieczorkiewicz M, Iwińska B, Kulczycki J, Gut W. [Subacute sclerosing panencephalitis (SSPE) in Poland 1987-1989. Stage IV of the epidemiological studies]. Przegl Epidemiol 1990; 44 (04) 265-271
    PubMedGoogle Scholar
  • 20 Naruszewicz-Lesiuk D, Iwińska-Buksowioz B, Wieczorkiewicz M, Kulczycki J, Gut W. [Subacute sclerosing panencephalitis (SSPE) in Poland in the years 1993-1995. The sixth stage of epidemiological research]. Przegl Epidemiol 1997; 51 (04) 371-380
    PubMedGoogle Scholar
  • 21 Naruszewicz-Lesiuk D, Iwińska-Buksowicz B, Wieczorkiewicz M, Kulczycki J, Gut W. [Subacute sclerosing panencephalitis (SSPE) in Poland in 1996-1999. Phase VII of epidemiologic studies]. Neurol Neurochir Pol 2000; 34 (05) 877-885
    PubMedGoogle Scholar
  • 22 Miller C, Andrews N, Rush M, Munro H, Jin L, Miller E. The epidemiology of subacute sclerosing panencephalitis in England and Wales 1990-2002. Arch Dis Child 2004; 89 (12) 1145-1148
    CrossrefPubMedGoogle Scholar
  • 23 Kija E, Ndondo A, Spittal G, Hardie DR, Eley B, Wilmshurst JM. Subacute sclerosing panencephalitis in South African children following the measles outbreak between 2009 and 2011. S Afr Med J 2015; 105 (09) 713-718
    CrossrefPubMedGoogle Scholar
  • 24 The Australian Paediatric Surveillance Unit [Internet]. Subacute Sclerosing Panencephalitis [accessed 2018 Jan 22]. Accessed April 2, 2021 at: http://www.apsu.org.au/assets/past-studies/sspe.pdf
    PubMedGoogle Scholar
  • 25 Saha NC, MollahMah SN, Yasmin L, Anwar S. Clinical and investigation profile of subacute sclerosing panencephalitis (SSPE): an analysis of twenty cases. J Dhaka Med Coll 2008; 17 (02) 72-77
    CrossrefPubMedGoogle Scholar
  • 26 Malik MA, Saeed M, Qureshi AU, Ahmed N, Akram M. Predictors of clinical course of subacute sclerosing panencephalitis: experience at the Children's Hospital, Lahore. J Coll Physicians Surg Pak 2010; 20 (10) 671-674
    PubMedGoogle Scholar
  • 27 Akram M, Naz F, Malik A, Hamid H. Clinical profile of subacute sclerosing panencephalitis. J Coll Physicians Surg Pak 2008; 18 (08) 485-488
    PubMedGoogle Scholar
  • 28 Rafique A, Amjad N, Chand P. et al. Subacute sclerosing panencephalitis: clinical and demographic characteristics. J Coll Physicians Surg Pak 2014; 24 (08) 557-560
    PubMedGoogle Scholar
  • 29 Jafri SK, Kumar R, Ibrahim SH. Subacute sclerosing panencephalitis - current perspectives. Pediatric Health Med Ther 2018; 9: 67-71
    CrossrefPubMedGoogle Scholar
  • 30 Ip P, Chung BH, Wong VC, Chan KY. Subacute sclerosing panencephalitis in children: prevalence in South China. Pediatr Neurol 2004; 31 (01) 46-51
    CrossrefPubMedGoogle Scholar
  • 31 Gutierrez J, Issacson RS, Koppel BS. Subacute sclerosing panencephalitis: an update. Dev Med Child Neurol 2010; 52 (10) 901-907
    CrossrefPubMedGoogle Scholar
  • 32 Dyken PR. Neuroprogressive disease of post-infectious origin: a review of a resurging subacute sclerosing panencephalitis (SSPE). Ment Retard Dev Disabil Res Rev 2001; 7 (03) 217-225
    CrossrefPubMedGoogle Scholar
  • 33 World Health Organization [Internet]. Global vaccine safety: subacute sclerosing panencephalitis and measles vaccination. Wkly Epidemiol Rec 2006; 81: 13-20 . Available at: https://www.who.int/vaccine_safety/committee/topics/measles_sspe/Jan_2006/en/
    PubMedGoogle Scholar
  • 34 Sonia M, Lalit D, Shobha B. et al. Subacute sclerosing panencephalitis in a tertiary care centre in post measles vaccination era. J Commun Dis 2009; 41 (03) 161-167
    PubMedGoogle Scholar
  • 35 Jagtap SA, Nair MD, Kambale HJ. Subacute sclerosing panencephalitis: a clinical appraisal. Ann Indian Acad Neurol 2013; 16 (04) 631-633
    CrossrefPubMedGoogle Scholar
  • 36 Bansal N, Narayan R. Subacute sclerosing panencephalitis after measles vaccination and without clinical measles, vaccine induced or something else: a case report. Pediatr Infect Dis 2014; 6: 18-20
    PubMedGoogle Scholar
  • 37 Chatterjee A, Sarkar A, Mukhopadhyay J. An interesting case of SSPE without past measles and normal vaccination. Ind J Med Case Rep ISSN: 2319–3832(Online) 2013; 2 (01) 30-3
    PubMedGoogle Scholar
  • 38 Aulakh R, Tiwari A. Subacute sclerosing panencephalitis in a toddler: changing epidemiological trends. Case Rep Pediatr 2013; 2013: 341462 DOI: 10.1155/2013/341462.
    CrossrefPubMedGoogle Scholar
  • 39 Wardhane S, Oswal J, Srivastava K. Atypical presentation of SSPE with short onset latency. J Nepal Paediatr Soc 2015; 35 (01) 62-63
    CrossrefPubMedGoogle Scholar
  • 40 Kumar S, Gupta R, Khare S, Sharma S. Atypical subacute sclerosing panencephalitis with short onset latency. Indian Pediatr 2013; 50 (02) 244-245
    CrossrefPubMedGoogle Scholar
  • 41 Ibrahim SH, Amjad N, Saleem AF, Chand P, Rafique A, Humayun KN. The upsurge of SSPE--a reflection of national measles immunization status in Pakistan. J Trop Pediatr 2014; 60 (06) 449-453
    CrossrefPubMedGoogle Scholar
  • 42 Mishra B, Kakkar N, Ratho RK, Singhi P, Prabhakar S. Changing trend of SSPE over a period of ten years. Indian J Public Health 2005; 49 (04) 235-237
    PubMedGoogle Scholar
  • 43 Narain JP, Banerjee KB. Measles in India: epidemiology and control. Indian J Pediatr 1989; 56 (04) 463-472
    CrossrefPubMedGoogle Scholar
  • 44 Malshe N, Palkar S, Kulkarni R, Lalwani S, Mishra AC, Arankalle V. Early disappearance of maternal anti-measles, mumps, rubella, and varicella antibodies in Indian infants. Vaccine 2019; 37 (11) 1443-1448
    CrossrefPubMedGoogle Scholar
  • 45 Waaijenborg S, Hahné SJM, Mollema L. et al. Waning of maternal antibodies against measles, mumps, rubella, and varicella in communities with contrasting vaccination coverage. J Infect Dis 2013; 208 (01) 10-16
    CrossrefPubMedGoogle Scholar
  • 46 Leuridan E, Hens N, Hutse V, Ieven M, Aerts M, Van Damme P. Early waning of maternal measles antibodies in era of measles elimination: longitudinal study. BMJ 2010; 340: c1626
    CrossrefPubMedGoogle Scholar
  • 47 Hara T, Yamashita S, Aiba H. et al. Measles virus-specific T helper 1/T helper 2-cytokine production in subacute sclerosing panencephalitis. J Neurovirol 2000; 6 (02) 121-126
    CrossrefPubMedGoogle Scholar
  • 48 Aaby P, Andersen M, Sodemann M, Jakobsen M, Gomes J, Fernandes M. Reduced childhood mortality after standard measles vaccination at 4-8 months compared with 9-11 months of age. BMJ 1993; 307 (6915): 1308-1311
    CrossrefPubMedGoogle Scholar
  • 49 Lochlainn LN, de Gier B, van der Maas N. et al. Measles vaccination below 9 months of age: Systematic literature review and meta analyses of effects and safety. National Institute for Public Health and the Environment (RIVM), Netherlands [Internet]. 2015
    Google Scholar