Cerebral amyloidotic angiopathy represents the most frequent cause of lobar haematoma in young patients and represents 5-10% of the non-traumatic cerebral haemorrhages. In the present work, we describe one autoptic case of recurrent cerebral haemorrhages in a 58-year-old woman. Macroscopically in the brain multiple haemorragic areas were present in the right frontal pole, right frontal and temporo-parietal lobes with homolateral ventricular inundation. The histological, histochemical, immunohistochemical, ultrastructural and biomolecular investigations confirmed the presence of amyloid deposits in the middle-size and little-size cerebral arteries. We report, moreover, a novel mutation (Leu705Val) within the Abeta sequence of a AbetaPP in a family with autosomal dominant, recurrent intracerebral hemorrhages beginning in the sixth decade of life.