Abstract
BackgroundAnomalies of pyramidal tract decussation are rare phenomena that can be caused by ectodermal dysplasia. Herein, we describe a patient with ichthyosis who exhibited ipsilateral hemiparesis after stroke and whose neuroimaging results showed evidence of motor control being provided by the ipsilateral motor cortex.Case presentationA 24-year-old right-handed man presented with skin abnormalities, sudden-onset left hemiparesis, and dysarthria. He exhibited a mild-to-moderate left-sided weakness (grade 4 on the Medical Research Council scale). Magnetic resonance imaging revealed an acute infarct in the left corona radiata. Diffusion tensor imaging revealed uncrossed corticospinal tracts. Next-generation sequencing identified heterozygous FLG mutations. The patient was diagnosed with cerebral infarction and ichthyosis vulgaris and was treated with aspirin (100 mg/d). His symptoms gradually dissipated.ConclusionsThis case suggests that pyramidal decussation anomalies can be associated with ichthyosis. Patients with ichthyosis should therefore be evaluated for nerve involvement.
Highlights
ConclusionsThis case suggests that pyramidal decussation anomalies can be associated with ichthyosis
Anomalies of pyramidal tract decussation are rare phenomena that can be caused by ectodermal dysplasia
This case suggests that pyramidal decussation anomalies can be associated with ichthyosis
Summary
We identified uncrossed pyramidal tracts in a patient with clinically and genetically confirmed ichthyosis and there might be a pathophysiological association, becoming clinically relevant in certain scenarios, such as stroke.
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