Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
. 2020 Apr 6;20(1):120.
doi: 10.1186/s12883-020-01698-0.

Uncrossed corticospinal tracts in a patient with ichthyosis and hemiparesis: a case report

Affiliations

Uncrossed corticospinal tracts in a patient with ichthyosis and hemiparesis: a case report

Huijia Yang et al. BMC Neurol. .

Abstract

Background: Anomalies 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 presentation: A 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.

Conclusions: This case suggests that pyramidal decussation anomalies can be associated with ichthyosis. Patients with ichthyosis should therefore be evaluated for nerve involvement.

Keywords: DTI; Ihthyosis; Ipsilateral hemiparesis; Ischemic stroke; Uncrossed corticospinal tracts.

PubMed Disclaimer

Conflict of interest statement

The authors declare that they have no competing interests.

Figures

Fig. 1
Fig. 1
The skin on the patient’s shins was dry and scaly
Fig. 2
Fig. 2
a. Diffusion-weighted imaging revealed lesions with strong signal intensities and decreased apparent diffusion coefficients. b. Diffusion tensor imaging tractography revealed uncrossed corticospinal tracts. The red arrow shows the infarcted area in which the corticospinal tract was destroyed

Similar articles

References

    1. Weiller C, Chollet F, Friston KJ, et al. Functional reorganization of the brain in recovery from striatocapsular infarction in man. Ann Neurol. 1992;31:463–472. doi: 10.1002/ana.410310502. - DOI - PubMed
    1. Hosokawa S, Tsuji S, Uozumi T, et al. Ipsilateral hemiplegia caused by right internal capsule and thalamic hemorrhage: demonstration of predominant ipsilateral innervation of motor and sensory systems by MRI, MEP, and SEP. Neurology. 1996;46:1146–1149. doi: 10.1212/wnl.46.4.1146. - DOI - PubMed
    1. ten Donkelaar HJ, Lammens M, Wesseling P, et al. Development and malformations of the human pyramidal tract. J Neurol. 2004;251:1429–1442. doi: 10.1007/s00415-004-0653-3. - DOI - PubMed
    1. Ng AS, Sitoh YY, Zhao Y, et al. Ipsilateral stroke in a patient with horizontal gaze palsy with progressive scoliosis and a subcortical infarct. Stroke. 2011;42:e1–e3. doi: 10.1161/STROKEAHA.110.591271. - DOI - PubMed
    1. Kang K, Choi N. Ipsilateral hemiparesis and spontaneous horizontal nystagmus caused by middle cerebral artery territory infarct in a patient with agenesis of the corpus callosum. Neurol Sci. 2012;33:1165–1168. doi: 10.1007/s10072-011-0871-2. - DOI - PubMed