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ngly1_deficiency [2025/03/11 02:16] – created Scott Larsonngly1_deficiency [2025/04/18 20:40] (current) – external edit 127.0.0.1
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 ====== NGLY1 Deficiency ====== ====== NGLY1 Deficiency ======
 +  * Global developmental delay
 +  * Intellectual diability
 +  * Hypotonia
 +  * Hypo- or alacrima
  
 +====Eye Findings====
 +  * Hypolacrima (83%)
 +  * Incomplete or abnormal blink (73%)
 +  * Optic disc abnormalities (73%)
 +    * Peripapillary atrophy, cupping 
 +  * Corneal abnormalities typically found with dry eye
 +    * punctate epithelial erosions
 +    * pannus and neovascularization
 +  * Conjunctival injection (60%)
 +  * Strabismus (33%)
 +    * Exotropia > Esotropia
 +    * Restricted ocular movements
 +  * Iris abnormalities 
 +    * Reduced pupillary reactivity (47%)
 +    * Irregular iris (27%)
 +  * Nystagmus- rare
 +====Other Findings====
 +  * Increased liver transaminases
 +  * Polyneuropathy
 +====Etiology====
 +  * Autosomal Recessive
 +  * Loss-of-function variants of the NGLY1 encoding N-glycanase 1
 +    * an enzyme that removes the sugar moiety from proteins
 +    * cleaves N-glycans to generate deglycosylated proteins and oligosaccharides containing 1-amino-GlcNac
 +    * degrades misfolded proteins in the endoplasmic reticulum 
 +====Resources====
 +  * {{ ::ocular_feature_of_ngly1_deficiency_from_a_prospective_lingitudinal_cohort_2023.pdf |Frater CH et al. Ocular features of NGLY1 deficiency from a prospective longitudinal cohort. J AAPOS 2024;28:103925}}
 +
 +{{tag>syndrome}}