Combined central and peripheral demyelination in two siblings, immune mediated or genetic?
Moodley KAnandan A MoodleyStephanie EfthymiouHenry HouldenPierre L A BillVinod B PatelSimon RinaldiPublished in: Practical neurology (2024)
We report unusual cases of combined central and peripheral demyelination in two siblings related to pregnancy, each presenting with progressive tetraparesis and cranial nerve palsies. The elder sister had a relapsing-remitting course with optic nerve dysfunction and died during a relapse from respiratory insufficiency. The younger sister presented with disorientation and acute-onset limb and facial weakness. She responded well to corticosteroid therapy. Their clinical presentation, response to immunomodulatory therapy, nerve conduction studies, cerebrospinal fluid and histology supported an acquired demyelinating cause. Whole-exome sequencing identified variants in two genes not previously linked to this clinical phenotype. Serological tests for antibody-mediated demyelination were negative. Despite the undefined pathogenesis, these cases provide a platform to explore the confluence of genetic, immune and environmental factors in the context of acquired demyelination. We discuss the differential diagnosis and a diagnostic approach to such cases from the perspectives of neuroimmunology and neurogenetics.
Keyphrases
- multiple sclerosis
- genome wide
- optic nerve
- copy number
- cerebrospinal fluid
- liver failure
- intellectual disability
- disease activity
- peripheral nerve
- oxidative stress
- respiratory failure
- high throughput
- drug induced
- case report
- dna methylation
- chemotherapy induced
- autism spectrum disorder
- cell therapy
- pregnant women
- extracorporeal membrane oxygenation
- soft tissue