| Literature DB >> 28532646 |
Maura Gallo1, Francesca Frangipane1, Chiara Cupidi1, Matteo De Bartolo2, Sabina Turone2, Camilla Ferrari3, Benedetta Nacmias4, Giuliana Grimaldi5, Valentina Laganà1, Rosanna Colao1, Livia Bernardi1, Maria Anfossi1, Maria Elena Conidi1, Franca Vasso1, Sabrina Anna Maria Curcio1, Maria Mirabelli1, Nicoletta Smirne1, Giusi Torchia1, Maria Gabriella Muraca1, Gianfranco Puccio1, Raffaele Di Lorenzo1, Maristella Piccininni6, Andrea Tedde4, Raffaele Giovanni Maletta1, Sandro Sorbi3, Amalia Cecilia Bruni7.
Abstract
We identified the novel PSEN1 pathogenic mutation M84V in 3 patients belonging to a large kindred affected by autosomal dominant Alzheimer's disease (AD). The clinical phenotype was characterized by early onset dementia in 14 affected subjects over 3 generations. Detailed clinical, imaging and genetic assessment was performed. We highlighted the presence of unusual symptoms such as frontal executive syndrome, psychosis and spastic paraparesis in these patients. Spastic paraparesis has been reported in other PSEN1 mutations in adjacent codons, suggesting that the position of the genetic defect may affect the clinical expression, although this phenotype can occur in mutations throughout the whole PSEN1 gene. Brain magnetic resonance imaging showed diffuse cortical atrophy, but also atrophy of cerebellar lobules, mainly involving Crus I, in 2 patients without cerebellar motor deficits. These neuroimaging results were consistent with recent findings about the association between sporadic AD and distinct and circumscribed cerebellar atrophy. The present work acknowledged the novel PSEN1 pathogenic mutation M84V and might contribute to the ongoing debate about the involvement of cerebellum in AD.Entities:
Keywords: Alzheimer's disease; Cerebellum; M84V; Presenilin 1 mutation; Spastic paraparesis
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Year: 2017 PMID: 28532646 DOI: 10.1016/j.neurobiolaging.2017.04.017
Source DB: PubMed Journal: Neurobiol Aging ISSN: 0197-4580 Impact factor: 4.673