Literature DB >> 24556562

Sensory stimulus-sensitive drop attacks and basal ganglia calcification: new findings in a patient with FOLR1 deficiency.

Sandra P Toelle1, David Wille1, Bernhard Schmitt1, Ianina Scheer2, Beat Thöny3, Barbara Plecko1.   

Abstract

Loss-of-function mutations in the FOLR1 gene (MIM *136430), encoding the folate receptor alpha, impair cerebral folate transport and lead to a progressive neurometabolic disorder. We report on a 5-year-old boy with progressive ataxia, from the age of 2 years and 6 months, with myoclonic jerks, regression, and impressive myoclonic tonic spasms with drop attacks, which were partially provoked by touching his face or washing his hands. Delayed myelination and cerebellar atrophy on cranial MRI were important clues to the diagnosis of cerebral folate transport deficiency, which was confirmed by homozygosity for the known nonsense mutation p.R204X in the FOLR1 gene. Computed tomography taken after head injury revealed bilateral calcifications in the basal ganglia as a novel finding in a patient with FOLR1 mutation.

Entities:  

Keywords:  FOLR1; MTHF; drop-attacks; hypomyelination; myoclonus

Mesh:

Substances:

Year:  2014        PMID: 24556562     DOI: 10.1684/epd.2014.0629

Source DB:  PubMed          Journal:  Epileptic Disord        ISSN: 1294-9361            Impact factor:   1.819


  4 in total

1.  Folinic acid therapy in cerebral folate deficiency: marked improvement in an adult patient.

Authors:  Ivan Karin; Ingo Borggraefe; Claudia B Catarino; Christoph Kuhm; Konstanze Hoertnagel; Saskia Biskup; Thomas Opladen; Nenad Blau; Florian Heinen; Thomas Klopstock
Journal:  J Neurol       Date:  2017-01-04       Impact factor: 4.849

Review 2.  Brain Calcification and Movement Disorders.

Authors:  Vladimir S Kostić; Igor N Petrović
Journal:  Curr Neurol Neurosci Rep       Date:  2017-01       Impact factor: 5.081

3.  A likely pathogenic variant in the SLC20A2 gene presenting with progressive myoclonus.

Authors:  Simon Lamquet; Eliana M Ramos; Andrea Legati; Giovanni Coppola; Dimitri Hemelsoet; Olivier M Vanakker
Journal:  Ann Clin Transl Neurol       Date:  2019-02-01       Impact factor: 4.511

4.  First case report of cerebral folate deficiency caused by a novel mutation of FOLR1 gene in a Chinese patient.

Authors:  Ciliu Zhang; Xiaolu Deng; Yafei Wen; Fang He; Fei Yin; Jing Peng
Journal:  BMC Med Genet       Date:  2020-11-26       Impact factor: 2.103

  4 in total

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