We report the neuropsychological profile and the pattern of brain activity during reading tasks in a sample of familial dyslexics. We studied our subjects with an in-depth neuropsychological assessment and with functional neuroimaging (fMRI) during word and pseudoword reading and false font string observations (baseline condition). The neuropsychological assessment revealed that familial dyslexia, in both persistent and compensated forms, is often associated with deficits in verbal short-term memory, phonological awareness and automatization abilities. The functional results showed a lack of activation in the posterior areas of the reading network. This study, together with the previously published VBM study (Brambati, S.M., Termine, C., Ruffino, M., Stella, G., Fazio, F., Cappa, S.F. and Perani, D., Regional reductions of gray matter volume in familial dyslexia, Neurology, 63 (2004) 742-5), provides a multiple modality evaluation of familial dyslexia. The neuropsychological assessment showed cognitive deficits associated with dyslexia that persist also in subjects with compensated reading deficit. Both the anatomical and the functional study point out a deficit in the posterior areas of the reading network.