| Literature DB >> 27982499 |
Anna Sagnelli1, Giuseppe Piscosquito1, Daniela Di Bella2, Laura Fadda3, Lisa Melzi4, Antonio Morico5, Claudia Ciano2, Franco Taroni2, Dante Facchetti6, Ettore Salsano1, Davide Pareyson1.
Abstract
We report the first Italian family affected by hereditary gelsolin amyloidosis (HGA), a rare autosomal dominant disease characterized by adult-onset slowly progressive cranial neuropathy, lattice corneal dystrophy, and cutis laxa. The index case was a 39-year-old male with a 9-year history of progressive bilateral facial nerve palsy. His mother had two episodes of acute facial palsy, and his maternal aunt and grandfather were also affected. Electrophysiological studies confirmed bilateral facial nerve involvement, without signs of peripheral polyneuropathy, and ophthalmological examination showed bilateral lattice corneal dystrophy, in both the index case and his mother. Gelsolin-gene sequencing revealed the heterozygous c.640G>A mutation (p.Asp187Asn) in the proband, his mother and aunt and also in three apparently asymptomatic relatives. The majority of HGA patients come from Finland, although several cases have been reported from other countries. HGA should be considered in the differential diagnosis of progressive or recurrent bilateral facial neuropathy.Entities:
Keywords: amyloidosis; bilateral facial palsy; familial amyloid polyneuropathy type IV; hereditary gelsolin amyloidosis; lattice corneal dystrophy
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Year: 2017 PMID: 27982499 DOI: 10.1111/jns.12200
Source DB: PubMed Journal: J Peripher Nerv Syst ISSN: 1085-9489 Impact factor: 3.494