Dopamine deficiency underlies learning deficits in neurofibromatosis-1 mice

Ann Neurol. 2013 Feb;73(2):309-15. doi: 10.1002/ana.23793. Epub 2012 Dec 7.

Abstract

Children with neurofibromatosis type 1 (NF1) are prone to learning and behavioral abnormalities, including problems with spatial learning and attention. The molecular etiology for these deficits is unclear, as previous studies have implicated defective dopamine, cyclic adenosine monophosphate (cAMP), and Ras homeostasis. Using behavioral, electrophysiological, and primary culture, we now demonstrate that reduced dopamine signaling is responsible for cAMP-dependent defects in neuron function and learning. Collectively, these results establish defective dopaminergic function as a contributing factor underlying impaired spatial learning and memory in children and adults with NF1, and support the use of treatments that restore normal dopamine homeostasis for select individuals.

Publication types

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

MeSH terms

  • Animals
  • Attention / physiology
  • Cyclic AMP / metabolism
  • Dopamine / deficiency*
  • Hippocampus / metabolism
  • Hippocampus / physiopathology
  • Learning Disabilities / etiology
  • Learning Disabilities / metabolism*
  • Learning Disabilities / physiopathology*
  • Memory Disorders / metabolism
  • Memory Disorders / physiopathology
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Neurofibromatosis 1 / complications
  • Neurofibromatosis 1 / metabolism*
  • Neurofibromatosis 1 / physiopathology*
  • Neurofibromin 1 / genetics
  • Signal Transduction / physiology
  • Ventral Tegmental Area / metabolism
  • Ventral Tegmental Area / physiopathology

Substances

  • Neurofibromin 1
  • Cyclic AMP
  • Dopamine