Resting-state fMRI functional connectome of C9orf72 mutation status

Ann Clin Transl Neurol. 2024 Mar;11(3):686-697. doi: 10.1002/acn3.51989. Epub 2024 Jan 17.

Abstract

Objective: The resting-state functional connectome has not been extensively investigated in amyotrophic lateral sclerosis (ALS) spectrum disease, in particular in relationship with patients' genetic status.

Methods: Here we studied the network-to-network connectivity of 19 ALS patients carrying the C9orf72 hexanucleotide repeat expansion (C9orf72+), 19 ALS patients not affected by C9orf72 mutation (C9orf72-), and 19 ALS-mimic patients (ALSm) well-matched for demographic and clinical variables.

Results: When compared with ALSm, we observed greater connectivity of the default mode and frontoparietal networks with the visual network for C9orf72+ patients (P = 0.001). Moreover, the whole-connectome showed greater node degree (P < 0.001), while sensorimotor cortices resulted isolated in C9orf72+.

Interpretation: Our results suggest a crucial involvement of extra-motor functions in ALS spectrum disease. In particular, alterations of the visual cortex may have a pathogenic role in C9orf72-related ALS. The prominent feature of these patients would be increased visual system connectivity with the networks responsible of the functional balance between internal and external attention.

MeSH terms

  • Amyotrophic Lateral Sclerosis* / diagnostic imaging
  • Amyotrophic Lateral Sclerosis* / genetics
  • Amyotrophic Lateral Sclerosis* / pathology
  • C9orf72 Protein / genetics
  • Connectome*
  • DNA Repeat Expansion / genetics
  • Humans
  • Magnetic Resonance Imaging
  • Mutation
  • Proteins / genetics

Substances

  • C9orf72 Protein
  • Proteins
  • C9orf72 protein, human