A Case of ALG6-CDG with Explosive Onset of Intractable Epilepsy During Infancy

Child Neurol Open. 2023 Feb 2:10:2329048X231153781. doi: 10.1177/2329048X231153781. eCollection 2023 Jan-Dec.

Abstract

ALG6-CDG is a rare, but second most common, type 1 congenital disorder of glycosylation (CDG) caused by a defect in the α-1-3-glucosyltransferase (ALG6) enzyme in the N-glycan assembly pathway. Many mutations have been identified and inherited in an autosomal recessive pattern. There are less than 100 ALG6-CDG cases reported, all sharing the phenotype of hypotonia and developmental delay. The majority (perhaps >70%) have seizures, but a minority have intractable epilepsy or epileptic encephalopathy. We report the clinical course, EEG findings, and neuroimaging of a child found to have compound heterozygous alleles c.257 + 5G > A and c.680G > A (p.G227E) who developed explosive onset of intractable epilepsy and epileptic encephalopathy. Initially, CDG was not suspected due to its rarity and lack of multi-organ system involvement, but rapid whole exam sequence (8-day turnaround) revealed the specific diagnosis quickly.

Keywords: EEG; congenital disorders of glycosylation; developmental delay; epilepsy; epileptic encephalopathy; inborn errors of metabolism.

Publication types

  • Case Reports