DOI: 10.4103/ipcares.ipcares_135_24 ISSN: 2772-5170

Infantile Epileptic Spasms Syndrome in an Infant with PHACTR1 Mutation Triggered by Whole-Cell-Pertussis Vaccine: A Case Report with Review of Literature

Sreejana Sharma, Murchana Khound, Bipul Kumar Das, Jaya Shankar Kaushik

Abstract

Background:

Early-onset infantile epileptic spasm syndrome is a known feature of phosphatase and actin regulator 1 (PHACTR1) gene mutation. However, its association with whole-cell pertussis vaccination is unknown.

Clinical Description:

A 1-year-old child presented with history of recurrent seizures for the past 8 months, having onset within a few hours after third dose of pentavalent vaccination containing whole-cell pertussis vaccine. There was associated developmental delay. Examination revealed generalized hypotonia with preserved reflexes.

Management and Outcome:

Routine blood investigations were largely normal. Electroencephalography showed classical hypsarrhythmia, while magnetic resonance imaging of the brain was unremarkable. Whole exome sequencing showed c.1499T>C heterozygous mutation in exon 12 of the PHACTR1 gene. The infant was treated with prednisolone, followed by vigabatrin, resulting in seizure-free state.

Conclusion:

Genetic analysis is advisable for infants with epileptic infantile spasms which are triggered by vaccination. PHACTR1 gene mutation could be one of the genetic variations, predisposing infants to vaccine-triggered seizures.

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