Subacute Sclerosing Panencephalitis in a Child Suffering from Human Immunodeficiency Virus on “Highly Active Antiretroviral Therapy” – Can This be Another Instance of Immune Reconstitution Inflammatory Syndrome?

Gupta, Ashutosh; Kushwaha, Suman; Manzoor, Mushbiq; Tarfarosh, Shah Faisal Ahmad

doi:10.7759/cureus.1346

Cited by 2 publications

(3 citation statements)

References 12 publications

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“…Also, as SMME has occurred in severely immunosuppressed HIV individuals, fulminant SSPE has been reported in HIV-infected individuals with good CD4 counts ( Sivadasan et al, 2012 ; Maurya et al, 2016 , Vidhale et al, 2021 ). It has been hypothesized that fulminant SSPE may be another example of an inflammatory immune reconstitution syndrome as a paradoxical exacerbation of a latent infection with mutant MV ( Gupta et al, 2017 ).…”

Section: Discussionmentioning

confidence: 99%

Subacute myoclonic measles encephalitis – An opportunistic HIV-associated infection

Ene

Duiculescu

Rădoi

et al. 2023

Front. Cell. Neurosci.

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IntroductionAn unusual cluster of myoclonic epilepsy was observed in a Romanian pediatric HIV cohort concurrent with measles outbreaks. We describe this particular form of subacute measles encephalitis (SME) in a group of HIV-infected children and adolescents with severe immunosuppression.MethodsThis is a single-center study, starting in 1997 and covering 4 measles outbreaks in Romania. The presumptive diagnosis of subacute myoclonic measles encephalitis (SMME) was based on: (1) epidemiological data, previous measles episode or presumed contact with measles virus (MV), (2) clinical presentation with initial localized myoclonic jerks with rapid extension and subsequent motor deficit with preserved mental status, and (3) neuroimaging studies revealing cortical gray matter lesions. Definitive diagnosis was based on a neuropathological exam and immunohistochemistry of brain tissues, and measles RNA detection in the cerebrospinal fluid (CSF).ResultsThirty-six patients were diagnosed with a particular form of SME during consecutive measles outbreaks in Romania: 1996–1998 (22); 2005–2008 (12); 2010–2011 (1) and 2016-2018 (1). Most children were born in the late 80s and had parenterally acquired HIV infection in early childhood. Before the episode of SMME, 11 patients had confirmed measles, while the rest, without typical rash, had a respiratory tract infection and/or presumed previous measles contact. In all patients, the clinical onset was sudden, with unilateral myoclonus. MRI findings revealed mainly focal cortical gray matter lesions. Neurologic symptoms progressed rapidly to coma and death in most patients. Three patients survived SMME, they had higher CD4 count at onset, slower progression of neurological symptoms, and benefit of immune recovery with cART. Immunocytochemistry studies revealed MV in the brain with a pattern suggesting an ascending viral neural infection. MV was isolated from CSF in 7 out of 8 patients. Sequence analysis of MV RNA from both nasopharyngeal swabs and CSF was available for one patient with similar N-450 strain characteristics.ConclusionDuring an outbreak of measles, neurological manifestations, especially myoclonus in immunosuppressed patients, can be related to measles even in the absence of an acute episode. This particular form of subacute myoclonic measles encephalitis is an opportunistic fatal disease. Immune recovery due to effective antiretroviral treatment might increase survival.

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Section: Discussionmentioning

confidence: 99%

Subacute myoclonic measles encephalitis – An opportunistic HIV-associated infection

Ene

Duiculescu

Rădoi

et al. 2023

Front. Cell. Neurosci.

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“…HIV impacts the immune system that can eventually affect the course of SSPE. Anti‐retroviral therapy induced changes in the immunological status has also been postulated to be a cause for its fulminant course 14‐16 …”

Section: Introductionmentioning

confidence: 99%

“…Anti-retroviral therapy induced changes in the immunological status has also been postulated to be a cause for its fulminant course. [14][15][16] Variations in interferon and cytokine responses, as well as cellmediated immunity, might attribute to the cause of the disease; in addition, data is sparse on the virus's genetic variations and the contribution of stage of the disease or the patient's genetic background. As a result, drawing conclusions is challenging.…”

Section: Introductionmentioning

confidence: 99%

Immune‐dysregulation in subacute sclerosing panencephalitis: An exploratory case–control study

Vijayvarman

Malhotra

Rizvi

et al. 2023

Journal of Medical Virology

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Subacute sclerosing panencephalitis (SSPE) is a chronic progressive neurological condition caused by a defective measles virus. It is postulated that immune‐dysregulation might result in persistent infection (immune evasion) as well as initiation of autoimmune phenomenon (via natural killer cells) leading to panencephalitis. The primary objective of this case‐control study was to analyse the pattern of immune dysregulation in cases with SSPE. The secondary objective was to assess the correlation between the measured immunological variables and disability/death at 6 months. This was an exploratory case–control study conducted at a tertiary‐care referral‐facility from January 2020 to September 2021. Thirty consecutive patients fulfilling the Dyken's criteria for SSPE and 30 age‐and‐sex‐matched healthy controls were enrolled. Immunological profile constituted by lymphocyte subset analysis, immunoglobulin levels and complement levels were done in all cases and controls. Cases were staged as per Jabbour's system; disability was assessed using the modified Rankin Scale (mRS). Patients with SSPE had a mean age of 14.76 years (±6.9 years). There were 25 males and 5 females; 6.7% cases belonged to Jabbour's first stage, 40% to second stage and 53.3% to third stage. At least 1/4th had evidence of measles vaccination. Levels of absolute lymphocyte count, B‐cells, T cells, helper T‐cells, and cytotoxic T‐cells were significantly higher in cases. IgG, IgM, and IgE levels were significantly higher while IgD levels were significantly lower in cases. At baseline, 13.3% of cases had a mRS score of 0–2 and 86.7% had a score of 3–6; at 6 months 10% had a mRS score 0–2 (favorable outcome) while 90% had a mRS score 3–6 (poor outcome). Higher IgE levels were found to correlate significantly with favorable outcome. Immune‐dysregulation may play a significant role in shaping one's response to measles infection as well as in determining vaccine‐efficacy.

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Subacute Sclerosing Panencephalitis in a Child Suffering from Human Immunodeficiency Virus on “Highly Active Antiretroviral Therapy” – Can This be Another Instance of Immune Reconstitution Inflammatory Syndrome?

Cited by 2 publications

References 12 publications

Subacute myoclonic measles encephalitis – An opportunistic HIV-associated infection

Subacute myoclonic measles encephalitis – An opportunistic HIV-associated infection

Immune‐dysregulation in subacute sclerosing panencephalitis: An exploratory case–control study

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