Sequential Presentation of Obsessive-Compulsive Disorder and Narcolepsy in a 10-Year-Old Girl With Wolfram Syndrome 1.
Rajith K RavindrenRajesh Thaliyil VeettilShibila AthimannilNeetha BalaramPrasad Thotton VeeduSajin Appadam VeetilArun Kumar AyoorSuja MathewKrishnakumar PadinharathShabeesh BalanPublished in: The Journal of nervous and mental disease (2024)
Wolfram syndrome 1 (WS1) is a rare, autosomal recessive neurodegenerative disorder characterized by diabetes insipidus, insulin-dependent diabetes mellitus, optic atrophy, and deafness resulting from loss-of-function genetic variants in the WFS1 gene. Individuals with WS1 manifest a spectrum of neuropsychiatric disorders. Here, we report a pediatric case of WS1, which stemmed from a novel biallelic WFS1 loss-of-function genetic variant. The individual initially presented with obsessive-compulsive disorder, which was successfully managed by fluvoxamine. After 2 months, the child manifested excessive daytime sleepiness. Clinical evaluation and sleep recordings revealed a diagnosis of narcolepsy type 2. Excessive daytime sleepiness was improved with methylphenidate. To the best of our knowledge, this is the first report of narcolepsy in WS1, which possibly arose during a progressive neurodegenerative process. We emphasize the need for in-depth screening for neuropsychiatric phenotypes and sleep-related disorders in WS1, for clinical management, which significantly improves the quality of life.
Keyphrases
- obsessive compulsive disorder
- sleep quality
- obstructive sleep apnea
- deep brain stimulation
- clinical evaluation
- type diabetes
- depressive symptoms
- glycemic control
- case report
- physical activity
- genome wide
- positive airway pressure
- weight gain
- copy number
- optical coherence tomography
- healthcare
- sleep apnea
- intellectual disability
- multiple sclerosis
- single cell
- metabolic syndrome
- dna methylation
- gene expression
- young adults
- optic nerve
- autism spectrum disorder
- skeletal muscle