Literature DB >> 28502191

A novel mutation in ALS2 associated with severe and progressive infantile onset of spastic paralysis.

Huma Tariq1, Shahid Mukhtar2, Sadaf Naz1.   

Abstract

Infantile onset ascending spastic paralysis (IAHSP) is a type of recessively inherited spastic paraplegia. We investigated the clinical and genetic cause of a recessively inherited disorder in two siblings manifesting severe spasticity in the lower limbs which hindered their gait. A novel homozygous nonsense mutation c.1918 C > T (p.Arg640*) was identified after whole-exome sequencing within ALS2 in the DNA of both patients. The obligate carriers were heterozygous for the mutation and other unaffected members were homozygous for the wild type allele. The variant was absent from 100 control chromosomes and all public databases. This report extends the allelic heterogeneity of ALS2 mutations and emphasizes the importance of genetic testing for diagnosis of pediatric disorders.

Entities:  

Keywords:  ALS2; Pakistan; Spastic paraplegia; alsin; infantile onset ascending spastic paralysis

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Substances:

Year:  2017        PMID: 28502191     DOI: 10.1080/01677063.2017.1324441

Source DB:  PubMed          Journal:  J Neurogenet        ISSN: 0167-7063            Impact factor:   1.250


  2 in total

1.  Clinical presentation and natural history of infantile-onset ascending spastic paralysis from three families with an ALS2 founder variant.

Authors:  Mayada Helal; Neda Mazaheri; Bita Shalbafan; Reza Azizi Malamiri; Nafi Dilaver; Rebecca Buchert; Javad Mohammadiasl; Neda Golchin; Alireza Sedaghat; Mohammad Yahya Vahidi Mehrjardi; Tobias B Haack; Olaf Riess; Wendy K Chung; Hamid Galehdari; Gholamreza Shariati; Reza Maroofian
Journal:  Neurol Sci       Date:  2018-08-21       Impact factor: 3.307

2.  Altered oligomeric states in pathogenic ALS2 variants associated with juvenile motor neuron diseases cause loss of ALS2-mediated endosomal function.

Authors:  Kai Sato; Asako Otomo; Mahoko Takahashi Ueda; Yui Hiratsuka; Kyoko Suzuki-Utsunomiya; Junya Sugiyama; Shuji Murakoshi; Shun Mitsui; Suzuka Ono; So Nakagawa; Hui-Fang Shang; Shinji Hadano
Journal:  J Biol Chem       Date:  2018-09-17       Impact factor: 5.157

  2 in total

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