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A juvenile ALS‐like phenotype dramatically improved after high‐dose riboflavin treatment

Christophe Carreau, Timothée Lenglet, Isabelle Mösnier, Ghizlène Lahlou, Guillaume Fargeot, Nicolas Weiss, Sophie Demeret, François Salachas, Alice Veauville‐Merllié, Cécile Acquaviva, Yann Nadjar

2020Annals of Clinical and Translational Neurology27 citationsDOIOpen Access PDF

Abstract

Riboflavin transporter deficiency (RTD) was recently characterized as a cause of genetic recessive childhood-onset motor neuron disease (MND) with hearing loss, formerly described as Brown-Vialetto-Van-Lear syndrome. We describe a 18-year-old woman with probable RTD mimicking juvenile Amyotrophic Lateral Sclerosis (ALS) who presented with an inaugural respiratory failure and moderate distal four limbs weakness. Only one heterozygous SLC52A3 mutation was detected, but presence of a sub-clinical auditory neuropathy and dramatic improvement under high dose riboflavin argued for a RTD. As RTD probably has a larger phenotypic spectrum than expected, a high dose riboflavin trial should be discussed in young-onset MND.

Topics & Concepts

RiboflavinAmyotrophic lateral sclerosisJuvenilePhenotypeWeaknessMyopathyMedicineRespiratory failurePediatricsGeneticsInternal medicineBiologyDiseaseSurgeryGeneBiochemistryMetabolism and Genetic DisordersRNA and protein synthesis mechanismsMitochondrial Function and Pathology
A juvenile ALS‐like phenotype dramatically improved after high‐dose riboflavin treatment | Litcius