Pigmentary retinal dystrophy associated with peroxisome biogenesis disorder-Zellweger syndrome spectrum.
Henry ZouLiliya SutherlandBrooke GeddiePublished in: Oxford medical case reports (2024)
Pigmentary retinal dystrophy (PRD) is a group of inherited disorders involving the progressive degeneration of rod and cone photoreceptors and the retinal pigment epithelium (RPE), which can progress to pigmentary retinopathy (PR). We present a case of PRD in a female pediatric patient who has pathogenic variants in the PRPH2 and PEX1 genes. The patient has associated macular edema and secondary visual impairment. Treatment has included serial dexamethasone intravitreal implant injections and topical dorzolamide. The PEX1 gene mutation is associated with peroxisome biogenesis disorder-Zellweger syndrome spectrum (PBD-ZSS) and resulting retinal dystrophies. The PRPH2 mutation may play a role in macular edema and PRD, as it is implicated in macular degeneration, choroid defects, and photoreceptor dysfunction. In this case, we review multiple gene mutations playing potential etiologic roles for PRD and discuss care management.
Keyphrases
- diabetic retinopathy
- optical coherence tomography
- case report
- early onset
- healthcare
- multiple sclerosis
- optic nerve
- oxidative stress
- palliative care
- genome wide
- low dose
- age related macular degeneration
- high dose
- copy number
- soft tissue
- dna methylation
- vascular endothelial growth factor
- platelet rich plasma
- risk assessment
- ultrasound guided
- pain management
- cataract surgery