Literature DB >> 29961494

The First Case of Riboflavin Transporter Deficiency in sub-Saharan Africa.

Shaakira Chaya1, Marco Zampoli2, Diane Gray2, Jane Booth2, Gillian Riordan3, Alvin Ndondo3, Karen Fieggen4, Jody Rusch5, George van der Watt5, Komala Pillay6, Francois van der Westhuizen7, Manoj Menezes8, Jo Wilmshurst3.   

Abstract

This report describes the first case of a child with genetically confirmed Brown-Vialetto-van Laere syndrome in sub-Saharan Africa. This is an extremely rare clinical condition that presents with an auditory neuropathy, bulbar palsy, stridor, muscle weakness, and respiratory compromise that manifests with diaphragmatic and vocal cord paralysis. It is an autosomal recessive condition for which the genetic mutation has only recently been linked to a riboflavin transporter deficiency. We describe an 11-month-old affected male infant. He has required long-term respiratory support and a gastrostomy tube to support feeding. With high-dose riboflavin supplementation, he had limited recovery of motor function. His respiratory chain enzyme studies were abnormal suggestive of mitochondrial (mt) dysfunction. In the setting of limited resources, recognition of this striking clinical phenotype is important to highlight, specifically regarding the genetic implications of the condition and the potentially remedial response to vitamin supplementation.
Copyright © 2017 Elsevier Inc. All rights reserved.

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Year:  2017        PMID: 29961494     DOI: 10.1016/j.spen.2017.03.002

Source DB:  PubMed          Journal:  Semin Pediatr Neurol        ISSN: 1071-9091            Impact factor:   1.636


  11 in total

1.  Brown-Vialetto-Van Laere syndrome: a novel diagnosis to a common presentation.

Authors:  Qalab Abbas; Sidra Kaleem Jafri; Sidra Ishaque; Arshalooz Jamila Rahman
Journal:  BMJ Case Rep       Date:  2018-06-27

Review 2.  Alteration of Flavin Cofactor Homeostasis in Human Neuromuscular Pathologies.

Authors:  Maria Tolomeo; Alessia Nisco; Maria Barile
Journal:  Methods Mol Biol       Date:  2021

3.  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

4.  To Be or No B2: A Rare Cause of Stridor and Weakness in a Toddler.

Authors:  Aliya L Frederick; Jennifer H Yang; Sarah Schneider; Alexis Quade; Lucia Guidugli; Kristen Wigby; Melissa Cameron
Journal:  Child Neurol Open       Date:  2021-08-05

5.  Hearing Impairment in South Africa and the Lessons Learned for Planetary Health Genomics: A Systematic Review.

Authors:  Noluthando Manyisa; Samuel Mawuli Adadey; Edmond Wonkam-Tingang; Abdoulaye Yalcouye; Ambroise Wonkam
Journal:  OMICS       Date:  2022-01

Review 6.  Development of Novel Experimental Models to Study Flavoproteome Alterations in Human Neuromuscular Diseases: The Effect of Rf Therapy.

Authors:  Maria Tolomeo; Alessia Nisco; Piero Leone; Maria Barile
Journal:  Int J Mol Sci       Date:  2020-07-26       Impact factor: 5.923

Review 7.  Peripheral nerve disease secondary to systemic conditions in children.

Authors:  Jo M Wilmshurst; Robert A Ouvrier; Monique M Ryan
Journal:  Ther Adv Neurol Disord       Date:  2019-08-12       Impact factor: 6.570

Review 8.  Is auditory neuropathy an appropriate term? A systematic literature review on its aetiology and pathogenesis.

Authors:  Sandro Burdo; Federica Di Berardino; Gabriele Bruno
Journal:  Acta Otorhinolaryngol Ital       Date:  2021-11-26       Impact factor: 2.124

9.  Effect of riboflavin deficiency on development of the cerebral cortex in Slc52a3 knockout mice.

Authors:  Congyun Jin; Atsushi Yonezawa; Hiroki Yoshimatsu; Satoshi Imai; Madoka Koyanagi; Kaori Yamanishi; Shunsaku Nakagawa; Kotaro Itohara; Tomohiro Omura; Takayuki Nakagawa; Junya Nagai; Kazuo Matsubara
Journal:  Sci Rep       Date:  2020-10-28       Impact factor: 4.379

10.  Targeted Therapies for Hereditary Peripheral Neuropathies: Systematic Review and Steps Towards a 'treatabolome'.

Authors:  Matthew J Jennings; Angela Lochmüller; Antonio Atalaia; Rita Horvath
Journal:  J Neuromuscul Dis       Date:  2021
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