The longitudinal evaluation of familial frontotemporal dementia subjects protocol: Framework and methodology

Alzheimers Dement. 2020 Jan;16(1):22-36. doi: 10.1016/j.jalz.2019.06.4947. Epub 2020 Jan 6.

Abstract

Introduction: It is important to establish the natural history of familial frontotemporal lobar degeneration (f-FTLD) and provide clinical and biomarker data for planning these studies, particularly in the asymptomatic phase.

Methods: The Longitudinal Evaluation of Familial Frontotemporal Dementia Subjects protocol was designed to enroll and follow at least 300 subjects for more than at least three annual visits who are members of kindreds with a mutation in one of the three most common f-FTLD genes-microtubule-associated protein tau, progranulin, or chromosome 9 open reading frame 72.

Results: We present the theoretical considerations of f-FTLD and the aims/objectives of this protocol. We also describe the design and methodology for evaluating and rating subjects, in which detailed clinical and neuropsychological assessments are performed, biofluid samples are collected, and magnetic resonance imaging scans are performed using a standard protocol.

Discussion: These data and samples, which are available to interested investigators worldwide, will facilitate planning for upcoming disease-modifying therapeutic trials in f-FTLD.

Keywords: C9orf72; Frontotemporal dementia; GRN; Genetics; MAPT; TDP-43; Tau.

Publication types

  • Research Support, N.I.H., Extramural

MeSH terms

  • Adult
  • C9orf72 Protein / genetics
  • Female
  • Frontotemporal Dementia / blood
  • Frontotemporal Dementia / cerebrospinal fluid
  • Frontotemporal Dementia / diagnostic imaging*
  • Frontotemporal Dementia / genetics*
  • Genetic Predisposition to Disease*
  • Humans
  • Longitudinal Studies
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Mutation / genetics
  • Neuropsychological Tests / statistics & numerical data*
  • tau Proteins / genetics

Substances

  • C9orf72 Protein
  • C9orf72 protein, human
  • MAPT protein, human
  • tau Proteins