Extensive genetics of ALS: a population-based study in Italy

Neurology. 2012 Nov 6;79(19):1983-9. doi: 10.1212/WNL.0b013e3182735d36. Epub 2012 Oct 24.

Abstract

Objective: To assess the frequency and clinical characteristics of patients with mutations of major amyotrophic lateral sclerosis (ALS) genes in a prospectively ascertained, population-based epidemiologic series of cases.

Methods: The study population includes all ALS cases diagnosed in Piemonte, Italy, from January 2007 to June 2011. Mutations of SOD1, TARDBP, ANG, FUS, OPTN, and C9ORF72 have been assessed.

Results: Out of the 475 patients included in the study, 51 (10.7%) carried a mutation of an ALS-related gene (C9ORF72, 32; SOD1, 10; TARDBP, 7; FUS, 1; OPTN, 1; ANG, none). A positive family history for ALS or frontotemporal dementia (FTD) was found in 46 (9.7%) patients. Thirty-one (67.4%) of the 46 familial cases and 20 (4.7%) of the 429 sporadic cases had a genetic mutation. According to logistic regression modeling, besides a positive family history for ALS or FTD, the chance to carry a genetic mutation was related to the presence of comorbid FTD (odds ratio 3.5; p = 0.001), and age at onset ≤54 years (odds ratio 1.79; p = 0.012).

Conclusions: We have found that ∼11% of patients with ALS carry a genetic mutation, with C9ORF72 being the commonest genetic alteration. Comorbid FTD or a young age at onset are strong indicators of a possible genetic origin of the disease.

Publication types

  • Research Support, N.I.H., Intramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Amyotrophic Lateral Sclerosis / epidemiology*
  • Amyotrophic Lateral Sclerosis / genetics*
  • C9orf72 Protein
  • Cell Cycle Proteins
  • Community Health Planning
  • DNA Mutational Analysis
  • DNA-Binding Proteins / genetics
  • Family Health
  • Frontotemporal Dementia / epidemiology
  • Frontotemporal Dementia / genetics
  • Gene Frequency
  • Genetic Predisposition to Disease / genetics*
  • Genotype
  • Humans
  • Italy / epidemiology
  • Membrane Transport Proteins
  • Middle Aged
  • Mutation / genetics*
  • Neuropsychological Tests
  • Proteins / genetics*
  • RNA-Binding Protein FUS / genetics
  • Retrospective Studies
  • Ribonuclease, Pancreatic / genetics
  • Superoxide Dismutase / genetics
  • Superoxide Dismutase-1
  • Transcription Factor TFIIIA / genetics

Substances

  • C9orf72 Protein
  • C9orf72 protein, human
  • Cell Cycle Proteins
  • DNA-Binding Proteins
  • Membrane Transport Proteins
  • OPTN protein, human
  • Proteins
  • RNA-Binding Protein FUS
  • SOD1 protein, human
  • Transcription Factor TFIIIA
  • Superoxide Dismutase
  • Superoxide Dismutase-1
  • angiogenin
  • Ribonuclease, Pancreatic