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Obstetric outcomes in pregnant women with subacute sclerosing panencephalitis (SSPE): a systematic review of case reports and case series

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Abstract

Background

Subacute sclerosing panencephalitis (SSPE) is a devastating brain disease caused by persistent infection by the measles virus. Several cases of SSPE in pregnant ladies have been described. This systematic review is focused on maternal and foetal outcomes among pregnant women with SSPE.

Methods

We searched four databases (PubMed, Embase, Scopus, and Google Scholar). We reviewed all relevant cases, published until 14 August 2022. The review was conducted following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. The protocol was registered with PROSPERO (CRD42022348630). The search items that we used were “((Pregnancy) OR (delivery)) AND (Subacute sclerosing panencephalitis (SSPE))”. Dyken’s criteria were used for the diagnosis of SSPE in pregnant women. The extracted data was recorded in an Excel sheet. The Joanna Briggs Institute Critical Appraisal tool for case reports was used to assess the quality of published cases.

Results

We came across 19 reports describing details of 21 cases. The age of SSPE-affected women varied from 14 to 34 years (mean 23 years). In the majority (n=14), clinical manifestations were started in the antepartum period. Nine pregnant SSPE women presented with vision loss. After delivery, 13 SSPE-affected women died. On the contrary, 15 foetuses, though the majority were preterm, were alive. Five foetuses either died soon after birth or were still-born.

Conclusion

In conclusion, SSPE in pregnancy is often missed, as it mimics eclampsia. SSPE in pregnancy usually has a devastating course. Universal early childhood measles vaccination is the only way to fight this menace.

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References

  1. Garg RK, Mahadevan A, Malhotra HS, Rizvi I, Kumar N, Uniyal R (2019) Subacute sclerosing panencephalitis. Rev Med Virol 29(5):e2058. https://doi.org/10.1002/rmv.2058

    Article  PubMed  Google Scholar 

  2. Garg RK, Paliwal V, Imran Rizvi, Shweta Pandey, Ravi Uniyal, Smriti Agrawal, Richa Khanna. 2022 Obstetric outcomes in pregnant women with subacute sclerosing panencephalitis (SSPE): a systematic review of case reports and case series. PROSPERO CRD42022348630 Available from: https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42022348630. Accessed 9 Aug 2022

  3. Dyken PR (1985) Subacute sclerosing panencephalitis. Current status. Neurol Clin 3(1):179–196

    Article  CAS  PubMed  Google Scholar 

  4. The Joanna Briggs Institute Critical Appraisal tools for use in JBI Systematic Reviews. Checklist for Case Reports. https://jbi.global/sites/default/files/2019-05/JBI_Critical_Appraisal-Checklist_for_Case_Reports2017_0.pd. Accessed 9 Aug 2022

  5. Nelson RF, Dennery JM, Montpetit V, Furesz (1972) J. S. S. P. E. and pregnancy. Lancet. 299(7763):1289. https://doi.org/10.1016/s0140-6736(72)91013-6.

  6. Gaines KJ, Jabbour JT, Whitaker JN, Sever J (1979) Subacute sclerosing panencephalitis during pregnancy. A surviving normal infant. Arch Neurol 36(5):314–316. https://doi.org/10.1001/archneur.1979.00500410092016

    Article  CAS  PubMed  Google Scholar 

  7. Wirguin I, Steiner I, Kidron D et al (1988) Fulminant subacute sclerosing panencephalitis in association with pregnancy. Arch Neurol 45(12):1324–1325. https://doi.org/10.1001/archneur.1988.00520360042009

    Article  CAS  PubMed  Google Scholar 

  8. Tan E, Namer IJ, Ciger A, Zileli T, Kucukali T (1991) The prognosis of subacute sclerosing panencephalitis in adults. Report of 8 cases and review of the literature. Clin Neurol Neurosurg 93(3):205–209. https://doi.org/10.1016/s0303-8467(05)80004-6

    Article  CAS  PubMed  Google Scholar 

  9. Mańko-Juraszek R, Kasperek S (1992) Podostre stwardniajace zapalenie mózgu (SSPE) w ciazy [Subacute sclerosing panencephalitis pregnancy]. Neurol Neurochir Pol 26(5):711–715

    PubMed  Google Scholar 

  10. Thiel A, Nau R, Fischer F et al (1996) Healthy infant delivered by a mother with subacute sclerosing panencephalitis during pregnancy. Neurology 47(6):1604. https://doi.org/10.1212/wnl.47.6.1604

    Article  CAS  PubMed  Google Scholar 

  11. Gascon GG, Frosch MP (1998) Case 15–1998. N Engl J Med 338(20):1448–1456. https://doi.org/10.1056/NEJM199805143382008

    Article  Google Scholar 

  12. Jayawant S, Feyi-Waboso A, Wallace S et al (2000) Retinitis and dementia in a pregnant girl: an unusual case. Eur J Paediatr Neurol 4(4):177–179. https://doi.org/10.1053/ejpn.2000.0296

    Article  CAS  PubMed  Google Scholar 

  13. Garg RK (2002) Subacute sclerosing panencephalitis. Postgrad Med J 78(916):63–70. https://doi.org/10.1136/pmj.78.916.63

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  14. Joseph FG, Jacob J, Carrington D (2002) SSPE - a forgotten disease? Eur J Neurol 9(5):542. https://doi.org/10.1046/j.1468-1331.2002.04452.x

    Article  CAS  PubMed  Google Scholar 

  15. Ortega-Aznar A, Romero-Vidal FJ, Castellví J, Ferrer JM, Codina A (2003) Adult-onset subacute sclerosing panencephalitis: clinico-pathological findings in 2 new cases. Clin Neuropathol 22(3):110–118

    CAS  PubMed  Google Scholar 

  16. Joseph F, Dawson K, Betmouni S et al (2005) The Bath Advanced Neurology Course 2003: progressive neurological decline in pregnancy. Practical Neurology 5:168–175

    Article  Google Scholar 

  17. Cole AJ, Henson JW, Roehrl MH, Frosch MP (2007) Case records of the Massachusetts General Hospital. Case 24-2007. A 20-year-old pregnant woman with altered mental status. N Engl J Med 357(6):589–600. https://doi.org/10.1056/NEJMcpc079018

    Article  CAS  PubMed  Google Scholar 

  18. Reis GF, Ritter JM, Bellini WJ, Rota PA, Bollen AW (2015) A 29-year-old pregnant woman with worsening left hemiparesis, encephalopathy, and hemodynamic instability: a case report of subacute sclerosing panencephalitis. Clin Neuropathol 34(5):258–266. https://doi.org/10.5414/NP300843

    Article  PubMed  Google Scholar 

  19. Rana M, Rana VM, Singh A, Singhal S (2015) Subacute sclerosing panencephalitis: a rare neurological disorder in pregnancy. J. Evol. Med. Dent. Sci 3(23):4033–4036. https://doi.org/10.14260/jemds/2015/580

    Article  Google Scholar 

  20. Nagar VS, Sawarkar R, Dhekne D, Hedau M, Kado R (2015) Subacute sclerosing panencephalitis in pregnancy. J Assoc Phy India 15:76–77

    Google Scholar 

  21. Chiu MH, Meatherall B, Nikolic A et al (2016) Subacute sclerosing panencephalitis in pregnancy. Lancet Infect Dis 16(3):366–375. https://doi.org/10.1016/S1473-3099(15)00524-1

    Article  PubMed  PubMed Central  Google Scholar 

  22. Sharma PK, Garg RK, Singla S (2020) Case report: subacute sclerosing panencephalitis in pregnancy mimicking posterior reversible encephalopathy syndrome. Am J Trop Med Hyg 102(3):634–636. https://doi.org/10.4269/ajtmh.19-0901

    Article  PubMed  PubMed Central  Google Scholar 

  23. Agarwal A, Sathish P, Salunkhe M et al (2021) Atypical subacute sclerosing panencephalitis (SSPE): all postpartum altered behavior isn’t CVT! Ann Indian Acad Neurol 24(6):1002–1004. https://doi.org/10.4103/aian.AIAN_1313_20

    Article  PubMed  PubMed Central  Google Scholar 

  24. World Health Organization. Subacute sclerosing panencephalitis and measles vaccination. Subacute sclerosing panencephalitis and measles vaccination. Extract from report of GACVS meeting of 1-2 December 2005, published in the WHO Weekly epidemiological Record on 13 January 2006.Down loaded from; https://www.who.int/groups/global-advisory-committee-on-vaccine-safety/topics/measles-vaccines#cms. Assessed on 20 August 2022

  25. Ambika S, Patil PS, Noronha V, Arjundas D, Bhende M, Majumdar PD (2022) Progressive vision loss and cognitive decline as presenting manifestation of adult onset subacute sclerosing panencephalitis. J Neuroophthalmol. https://doi.org/10.1097/WNO.0000000000001587

  26. Herath H, Nandasiri S, Chandrakumara A, Jayamaha J, Senanayake S, Goonaratne K (2020) Subacute sclerosing panencephalitis: a rare cause of acute cortical visual impairment in an adult. Trop Doct 50(4):354–358. https://doi.org/10.1177/0049475520939325

    Article  PubMed  Google Scholar 

  27. Barthwal S, Kaur N, Kaur R, Zaidi R, Randev S (2021) Magnetic resonance imaging in subacute sclerosing panencephalitis: two case reports and review of literature. Cureus 13(10):e19161. https://doi.org/10.7759/cureus.19161

    Article  PubMed  PubMed Central  Google Scholar 

  28. Risk WS, Haddad FS (1979) The variable natural history of subacute sclerosing panencephalitis: a study of 118 cases from the Middle East. Arch Neurol 36(10):610–614. https://doi.org/10.1001/archneur.1979.00500460044004

    Article  CAS  PubMed  Google Scholar 

  29. PeBenito R, Naqvi SH, Arca MM, Schubert R (1997) Fulminating subacute sclerosing panencephalitis: case report and literature review. Clin Pediatr (Phila) 36(3):149–154. https://doi.org/10.1177/000992289703600306

    Article  CAS  PubMed  Google Scholar 

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Correspondence to Ravindra Kumar Garg.

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Supplementary information

Supplementary file 1

Supplementary item-1: Search strategy, Supplementary item-2: Diagnostic criteria of SSPE, Supplementary item-3: PRISMA checklist, Supplementary item-4: The Joanna Briggs Institute Critical Appraisal tool for case reports.

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Garg, R.K., Paliwal, V., Rizvi, I. et al. Obstetric outcomes in pregnant women with subacute sclerosing panencephalitis (SSPE): a systematic review of case reports and case series. Neurol Sci 44, 1959–1968 (2023). https://doi.org/10.1007/s10072-023-06650-x

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