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Abstract
Multivitamin (Thiamine) Responsive Basal Ganglia Disease Mimicking Acute Encephalitis Syndrome among Infants of Bodo Tribe Assam: A Retrospective Study
Background: Bilateral symmetrical basal ganglia infarcts were observed among infants, who presented with features of Acute Encephalitis Syndrome (AES). Thiamine is successfully tried in the management of the neurological condition, with basal ganglia involvement. Here we are comparing the treatment and outcome differences between infants who received multivitamins (thiamine), with infants who received only supportive care.
Methodology: In this retrospective study, done from the hospital medical record of a secondary level hospital in Assam, Northeast India, between 2011-2015; 50 infants had bilateral basal ganglia infarcts, encompassing our study population. Depending on the exposure to multivitamins (thiamine), introduced in May 2014; 27 infants were grouped in the non-exposure group (Sep 2011-April 2014), and 23 infants were grouped in the exposure group (May 2014-Sep 2015).
Results: Common presenting symptoms included seizures (100%), lethargy (90%), fever (70%), and feeding difficulties (76%). In the exposure group 1 (3.7%) infant died and in the non-exposure group 20 infants (86.9%) died (relative risk, 0.04; 95% Confidence Interval [CI], 0.006 to 0.29; P=0.00013). The infants in the exposure group have 96% less risk of death, compared with the non-exposure group. Two infants in subsequent follow-up, in the exposure group, did not have any neurological sequelae.
Conclusions: Intravenous multivitamin (thiamine) supplementation may be associated with less risk of death in infants who had bilateral symmetrical basal ganglia infarct in the brain and presented with symptoms of Acute Encephalitis Syndrome (AES). The study suggests the possibility of subclinical thiamine deficiency, mitochondrial diseases, or SLC19A3 gene mutation in this population.
Published Date: 2021-09-30; Received Date: 2021-06-30