Literature DB >> 28444446

Comparison of the clinical and cognitive features of genetically positive ALS patients from the largest tertiary center in Serbia.

Ivan V Marjanović1, Biljana Selak-Djokić2, Stojan Perić2, Milena Janković2, Vladimir Arsenijević3, Ivana Basta4, Dragana Lavrnić4, Elka Stefanova4, Zorica Stević4.   

Abstract

Discovering novel mutations in C9orf72, FUS, ANG, and TDP-43 genes in ALS patients arises necessities for better clinical characterizations of these subjects. The aim is to determine clinical and cognitive profile of genetically positive Serbian ALS patients. 241 ALS patients were included in the study (17 familiar and 224 apparently sporadic). The following genes were analyzed: SOD1, C9orf72, ANG, FUS, and TDP-43. An extensive battery of classic neuropsychological tests was used in 27 ALS patients (22 SOD1 positive and 5 SOD1 negative) and 82 healthy controls (HCs). Overall 37 (15.4%) of 241 ALS patients carried mutations in tested genes-among 17 familiar ALS patients 16 (94.1%) were positive and among 224 apparently sporadic 21 (9.4%) had causative mutation. Mutations in SOD1 gene were the most common, representing 27 (73.0%) of all genetically positive ALS patients. The main clinical characteristics of SOD1 positive patients were: spinal onset in lower extremities, common sphincter and sensitive disturbances, and dysexecutive syndrome. Within SOD1 positive patients, we noticed somewhat earlier onset in patients with A145G, sensory and sphincter disturbances were dominant in patients with L144F, while D90A patients had significant sensory involvement. SOD1 negative group consisted of ten (27.0%) patients (six C9orf72, two ANG, one TDP-43, and one patient baring triple FUS, C9orf72 expansion, and ANG variants). Bulbar involvement and more extensive neuropsychological impairment (including executive, visuospatial, and memory difficulties) were the main features of SOD1 negative cohort. Our results suggest that meaningful clinical suspicion of certain ALS genotype might be made based on thorough clinical evaluation of patients.

Entities:  

Keywords:  ALS; C9orf72; Cognition; Genetics; SOD1

Mesh:

Substances:

Year:  2017        PMID: 28444446     DOI: 10.1007/s00415-017-8495-y

Source DB:  PubMed          Journal:  J Neurol        ISSN: 0340-5354            Impact factor:   4.849


  33 in total

Review 1.  El Escorial revisited: revised criteria for the diagnosis of amyotrophic lateral sclerosis.

Authors:  B R Brooks; R G Miller; M Swash; T L Munsat
Journal:  Amyotroph Lateral Scler Other Motor Neuron Disord       Date:  2000-12

2.  Expanded GGGGCC hexanucleotide repeat in noncoding region of C9ORF72 causes chromosome 9p-linked FTD and ALS.

Authors:  Mariely DeJesus-Hernandez; Ian R Mackenzie; Bradley F Boeve; Adam L Boxer; Matt Baker; Nicola J Rutherford; Alexandra M Nicholson; NiCole A Finch; Heather Flynn; Jennifer Adamson; Naomi Kouri; Aleksandra Wojtas; Pheth Sengdy; Ging-Yuek R Hsiung; Anna Karydas; William W Seeley; Keith A Josephs; Giovanni Coppola; Daniel H Geschwind; Zbigniew K Wszolek; Howard Feldman; David S Knopman; Ronald C Petersen; Bruce L Miller; Dennis W Dickson; Kevin B Boylan; Neill R Graff-Radford; Rosa Rademakers
Journal:  Neuron       Date:  2011-09-21       Impact factor: 17.173

3.  Genetic studies of Russian patients with amyotrophic lateral sclerosis.

Authors:  Elena V Lysogorskaia; Nataliya Yu Abramycheva; Mariya N Zakharova; Mariya S Stepanova; Anna A Moroz; Alexey V Rossokhin; Sergey N Illarioshkin
Journal:  Amyotroph Lateral Scler Frontotemporal Degener       Date:  2015-11-09       Impact factor: 4.092

4.  A case of ALS-FTD in a large FALS pedigree with a K17I ANG mutation.

Authors:  M A van Es; F P Diekstra; J H Veldink; F Baas; P R Bourque; H J Schelhaas; E Strengman; E A M Hennekam; D Lindhout; R A Ophoff; L H van den Berg
Journal:  Neurology       Date:  2009-01-20       Impact factor: 9.910

5.  A de novo missense mutation of the FUS gene in a "true" sporadic ALS case.

Authors:  Adriano Chiò; Andrea Calvo; Cristina Moglia; Irene Ossola; Maura Brunetti; Luca Sbaiz; Shiao-lin Lai; Yevgeniya Abramzon; Bryan J Traynor; Gabriella Restagno
Journal:  Neurobiol Aging       Date:  2010-07-03       Impact factor: 4.673

6.  Cognitive change in ALS: a prospective study.

Authors:  S Abrahams; P N Leigh; L H Goldstein
Journal:  Neurology       Date:  2005-04-12       Impact factor: 9.910

7.  Dementia and aphasia in motor neuron disease: an underrecognised association?

Authors:  W P Rakowicz; J R Hodges
Journal:  J Neurol Neurosurg Psychiatry       Date:  1998-12       Impact factor: 10.154

8.  TDP-43 mutation in familial amyotrophic lateral sclerosis.

Authors:  Akio Yokoseki; Atsushi Shiga; Chun-Feng Tan; Asako Tagawa; Hiroyuki Kaneko; Akihide Koyama; Hiroto Eguchi; Akira Tsujino; Takeshi Ikeuchi; Akiyoshi Kakita; Koichi Okamoto; Masatoyo Nishizawa; Hitoshi Takahashi; Osamu Onodera
Journal:  Ann Neurol       Date:  2008-04       Impact factor: 10.422

9.  G41S SOD1 mutation: A common ancestor for six ALS Italian families with an aggressive phenotype.

Authors:  Stefania Battistini; Claudia Ricci; Fabio Giannini; Silvia Calzavara; Giuseppe Greco; Alberto Del Corona; Michelangelo Mancuso; Noè Battistini; Gabriele Siciliano; Paola Carrera
Journal:  Amyotroph Lateral Scler       Date:  2010

10.  Genetic overlap between apparently sporadic motor neuron diseases.

Authors:  Marka van Blitterswijk; Lotte Vlam; Michael A van Es; W-Ludo van der Pol; Eric A M Hennekam; Dennis Dooijes; Helenius J Schelhaas; Anneke J van der Kooi; Marianne de Visser; Jan H Veldink; Leonard H van den Berg
Journal:  PLoS One       Date:  2012-11-14       Impact factor: 3.240

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