Literature DB >> 34384672

Three cases of adult-onset Brown-Vialetto-Van Laere syndrome: Novel variants in SLC52A3 gene and MRI abnormalities.

Guillaume Carey1, Gregory Kuchcinski2, Fanny Gauvrit3, Luc Defebvre4, Sylvie Nguyen5, Claire-Marie Dhaenens6, Anne Frédérique Dessein6, Christine Vianey-Saban7, Cécile Acquaviva7, Céline Tard8.   

Abstract

Brown-Vialetto-Van Laere syndrome is a rare, autosomal, recessive neurological condition caused by variants in the riboflavin transporter genes SLC52A2 and SLC52A3. Here, we report on three cases. Case 1 was a 35-year-old woman from a consanguineous family who presented with progressive deafness, subacute multiple cranial nerve impairments (III, VII, IX, XII), and MRI abnormalities (including as hypersignal from the cranial nerves). The patient was homozygous for a novel SLC52A3variant. Case 2 was the woman's brother, who presented similar symptoms. Case 3 was an 18-year-old woman experiencing progressive hearing loss, bilateral steppage gait and a cranial nerves impairment (VII and XII). MRI revealed hypersignal in the root nerves and cauda equina. A novel heterozygous variant in SLC52A3 was identified. A subacute history of polyradiculoneuropathy along with progressive deafness, cranial nerve impairment, and MRI abnormalities should raise suspicion for Brown-Vialetto-Van Laere syndrome.
Copyright © 2021 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  BVVL; Brown-Vialetto-Van Laere syndrome; MRI; SLC52A3; tongue fasciculation

Mesh:

Substances:

Year:  2021        PMID: 34384672     DOI: 10.1016/j.nmd.2021.06.009

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  1 in total

1.  Riboflavin deficiency leads to irreversible cellular changes in the RPE and disrupts retinal function through alterations in cellular metabolic homeostasis.

Authors:  Tirthankar Sinha; Larissa Ikelle; Mustafa S Makia; Ryan Crane; Xue Zhao; Mashal Kakakhel; Muayyad R Al-Ubaidi; Muna I Naash
Journal:  Redox Biol       Date:  2022-06-16       Impact factor: 10.787

  1 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.