Literature DB >> 27982499

Hereditary gelsolin amyloidosis (HGA): a neglected cause of bilateral progressive or recurrent facial palsy.

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.
© 2016 Peripheral Nerve Society.

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


  2 in total

1.  Facial Palsy, Radiographic and Other Workup Negative: FROWN.

Authors:  Jacqueline J Greene; Reza Sadjadi; Nate Jowett; Tessa Hadlock
Journal:  Neurol Clin Pract       Date:  2021-10

2.  Gelsolin pathogenic Gly167Arg mutation promotes domain-swap dimerization of the protein.

Authors:  Francesco Bonì; Mario Milani; Alberto Barbiroli; Luisa Diomede; Eloise Mastrangelo; Matteo de Rosa
Journal:  Hum Mol Genet       Date:  2018-01-01       Impact factor: 6.150

  2 in total

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