Literature DB >> 32281455

The Occurrence of FUS Mutations in Pediatric Amyotrophic Lateral Sclerosis: A Case Report and Review of the Literature.

Vincent Picher-Martel1,2,3, Francis Brunet1,3, Nicolas Dupré1, Nicolas Chrestian4.   

Abstract

Amyotrophic lateral sclerosis (ALS) is a progressive motor neuron disease affecting both upper and lower motor neurons and leading to progressive paralysis. Most cases are sporadic, and the symptoms generally begin in the sixth or seventh decade. Juvenile ALS appears in a rare subgroup of patients with onset before the age of 25 years old. Contrary to the classical adult phenotype where 90% of cases are sporadic, most cases of juvenile ALS are caused by a genetic mutation in either SOD1 (superoxide dismutase one), SETX (senataxin), or FUS (fused in sarcoma). In the pediatric population, ALS is more infrequent and rarely considered in the differential diagnosis. There are few reports of ALS in children. Here, we describe a 14-year-old boy with a very fast progressing classical ALS phenotype and tremor caused by a c.1554_1557delACAG mutation in FUS. Our review of the literature advocates that pediatric ALS is highly suggestive of FUS mutations and that gene should be tested in children presenting with symptoms of ALS. The children with FUS-related ALS may have no family history and present initially with learning disabilities, tremor, and mild motor developmental delay.

Entities:  

Keywords:  adolescents; developmental disability; genetics; mutation; pediatric

Year:  2020        PMID: 32281455     DOI: 10.1177/0883073820915099

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  6 in total

Review 1.  Approaches to Gene Modulation Therapy for ALS.

Authors:  Katharina E Meijboom; Robert H Brown
Journal:  Neurotherapeutics       Date:  2022-09-06       Impact factor: 6.088

2.  Evidence of synergism among three genetic variants in a patient with LMNA-related lipodystrophy and amyotrophic lateral sclerosis leading to a remarkable nuclear phenotype.

Authors:  Kathryn Volkening; Sali M K Farhan; Jessica Kao; Cheryl Leystra-Lantz; Lee Cyn Ang; Adam McIntyre; Jian Wang; Robert A Hegele; Michael J Strong
Journal:  Mol Cell Biochem       Date:  2021-03-04       Impact factor: 3.396

3.  Generation and analysis of innovative genomically humanized knockin SOD1, TARDBP (TDP-43), and FUS mouse models.

Authors:  Anny Devoy; Georgia Price; Francesca De Giorgio; Rosie Bunton-Stasyshyn; David Thompson; Samanta Gasco; Alasdair Allan; Gemma F Codner; Remya R Nair; Charlotte Tibbit; Ross McLeod; Zeinab Ali; Judith Noda; Alessandro Marrero-Gagliardi; José M Brito-Armas; Chloe Williams; Muhammet M Öztürk; Michelle Simon; Edward O'Neill; Sam Bryce-Smith; Jackie Harrison; Gemma Atkins; Silvia Corrochano; Michelle Stewart; Jonathan D Gilthorpe; Lydia Teboul; Abraham Acevedo-Arozena; Elizabeth M C Fisher; Thomas J Cunningham
Journal:  iScience       Date:  2021-11-15

Review 4.  Motor neuron-derived induced pluripotent stem cells as a drug screening platform for amyotrophic lateral sclerosis.

Authors:  Mariana A Amorós; Esther S Choi; Axel R Cofré; Nikolay V Dokholyan; Marcelo Duzzioni
Journal:  Front Cell Dev Biol       Date:  2022-08-24

5.  MRI-guided histology of TDP-43 knock-in mice implicates parvalbumin interneuron loss, impaired neurogenesis and aberrant neurodevelopment in amyotrophic lateral sclerosis-frontotemporal dementia.

Authors:  Ziqiang Lin; Eugene Kim; Mohi Ahmed; Gang Han; Camilla Simmons; Yushi Redhead; Jack Bartlett; Luis Emiliano Pena Altamira; Isobel Callaghan; Matthew A White; Nisha Singh; Stephen Sawiak; Tara Spires-Jones; Anthony C Vernon; Michael P Coleman; Jeremy Green; Christopher Henstridge; Jeffrey S Davies; Diana Cash; Jemeen Sreedharan
Journal:  Brain Commun       Date:  2021-05-27

Review 6.  Juvenile Amyotrophic Lateral Sclerosis: A Review.

Authors:  Tanya Lehky; Christopher Grunseich
Journal:  Genes (Basel)       Date:  2021-11-30       Impact factor: 4.096

  6 in total

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