Top

BMC Neurology

Published in:

01-12-2020 | Magnetic Resonance Imaging | Case report

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

Authors: Huijia Yang, Hongwei Zhou, Jing Miao

Published in: BMC Neurology | Issue 1/2020

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.

Weiller C, Chollet F, Friston KJ, et al. Functional reorganization of the brain in recovery from striatocapsular infarction in man. Ann Neurol. 1992;31:463–72. https://doi.org/10.1002/ana.410310502.CrossRefPubMed

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;46:1146–9. https://doi.org/10.1212/wnl.46.4.1146.CrossRefPubMed

ten Donkelaar HJ, Lammens M, Wesseling P, et al. Development and malformations of the human pyramidal tract. J Neurol. 2004;251:1429–42. 2005/01/13. https://doi.org/10.1007/s00415-004-0653-3.CrossRefPubMed

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:e1–3. 2010/11/23. https://doi.org/10.1161/STROKEAHA.110.591271.CrossRefPubMed

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;33:1165–8. 2011/12/14. https://doi.org/10.1007/s10072-011-0871-2.CrossRefPubMed

Zhang H, Guo Y, Wang W, et al. Mutations in the filaggrin gene in Han Chinese patients with atopic dermatitis. Allergy. 2011;66:420–7. 2010/11/03. https://doi.org/10.1111/j.1398-9995.2010.02493.x.CrossRefPubMed

Chen H, Common JE, Haines RL, et al. Wide spectrum of filaggrin-null mutations in atopic dermatitis highlights differences between Singaporean Chinese and European populations. Br J Dermatol. 2011;165:106–14. 2011/03/25. https://doi.org/10.1111/j.1365-2133.2011.10331.x.CrossRefPubMed

Paller AS. Profiling Immune Expression to Consider Repurposing Therapeutics for the Ichthyoses. J Invest Dermatol. 2019;139:535–40. 2019/01/24. https://doi.org/10.1016/j.jid.2018.08.027.CrossRefPubMed

Roy U, Das U, Pandit A, et al. Sjogren-Larsson syndrome: a rare disease of the skin and central nervous system. BMJ Case Rep 2016; 2016: 10 1136/bcr-2016-215110. 2016/04/21. DOI: https://doi.org/10.1136/bcr-2016-215110.

10.

Cincotta M, Borgheresi A, Liotta P, et al. Reorganization of the motor cortex in a patient with congenital hemiparesis and mirror movements. Neurology. 2000;55:129. https://doi.org/10.1212/WNL.55.1.129.CrossRefPubMed

11.

Terakawa H, Abe K, Nakamura M, et al. Ipsilateral hemiparesis after putaminal hemorrhage due to uncrossed pyramidal tract. Neurology. 2000;54:1801–5. https://doi.org/10.1212/wnl.54.9.1801.CrossRefPubMed

12.

Ueki Y, Mima T, Oga T, et al. Dominance of ipsilateral corticospinal pathway in congenital mirror movements. J Neurol Neurosurg Psychiatry. 2005;76:276–9. 2005/01/18. https://doi.org/10.1136/jnnp.2004.040949.CrossRefPubMedPubMedCentral

13.

Sprecher E, Ishida-Yamamoto A, Mizrahi-Koren M, et al. A mutation in SNAP29, coding for a SNARE protein involved in intracellular trafficking, causes a novel neurocutaneous syndrome characterized by cerebral dysgenesis, neuropathy, ichthyosis, and palmoplantar keratoderma. Am J Hum Genet. 2005;77:242–51. https://doi.org/10.1086/432556.CrossRefPubMedPubMedCentral

Title: Uncrossed corticospinal tracts in a patient with ichthyosis and hemiparesis: a case report
Authors: Huijia Yang
Hongwei Zhou
Jing Miao
Publication date: 01-12-2020
Publisher: BioMed Central
Keywords: Magnetic Resonance Imaging
Magnetic Resonance Imaging
Stroke
Ichthyosis
Published in: BMC Neurology / Issue 1/2020
Electronic ISSN: 1471-2377
DOI: https://doi.org/10.1186/s12883-020-01698-0

Keynote webinar | Spotlight on medication adherence

Springer Medicine

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

Abstract

Background

Case presentation

Conclusions

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Background

Case presentation

Conclusions

Please log in to get access to this content

Other articles of this Issue 1/2020

Tranexamic acid is associated with reduced mortality, hemorrhagic expansion, and vascular occlusive events in traumatic brain injury – meta-analysis of randomized controlled trials

Extrastriatal 123I-FP-CIT SPECT impairment in Parkinson’s disease – the PPMI cohort

Severe cerebral edema induced by watershed shift after bypass in a patient with chronic steno-occlusive disease: a case report and short literature review

The neuropsychological assessment battery (NAB) is a valuable tool for evaluating neuropsychological outcome after aneurysmatic subarachnoid hemorrhage

Hyperacute relapse of Lewis-Sumner syndrome during influenza A (H1N1) virus infection

Stakeholder perceptions of components of a Parkinson disease care management intervention, care coordination for health promotion and activities in Parkinson’s disease (CHAPS)