Literature DB >> 34207092

Ocular Involvement in Hereditary Transthyretin Amyloidosis: A Case Series Describing Novel Potential Biomarkers.

Angelo Maria Minnella1,2, Roberta Rissotto1,2, Martina Maceroni1, Angela Romano3, Romina Fasciani2, Marco Luigetti2,3, Mario Sabatelli3,4, Stanislao Rizzo1,2, Benedetto Falsini1,2.   

Abstract

Hereditary transthyretin amyloidosis (hATTR) is a rare disease caused by a point mutation in the transthyretin (TTR) gene and inherited in an autosomal dominant fashion. TTR is a plasma protein that functions as a carrier for thyroxine (T4) and retinol (vitamin A). Ophthalmological manifestations are due to both the hepatic and ocular production of mutated TTR. In this case series, we report the ocular manifestations of hATTR in eighteen eyes of nine consecutive patients. Corneal nerve abnormalities as well as morphological and functional changes in the retina were investigated. The study was a single-center, retrospective, observational, clinical case series. In all patients, corneal confocal microscopy (CCM), multimodal imaging of the retina, including fundus photography and Optical Coherence Tomography (OCT), as well as rod and cone electroretinography (ERG) were performed. Eight patients had active disease and one was an unaffected carrier. In all study eyes, corneal nerve plexa examined with CCM were poorly represented or absent. Mixed rod-cone and cone ERG b-wave amplitudes were reduced, and photopic b-wave responses were significantly delayed. Photopic Negative Response (PhNR) amplitude was significantly reduced, while PhNR latency was significantly augmented. In 13/18 eyes, vitreous opacities and abnormalities of vitreo-retinal interface were found. The current results highlight the presence of corneal nerve damage. Functional retinal abnormalities, detected by ERG, can be found even in the presence of minimal or absent structural retinal damage. These findings support the use of CCM and ERGs to detect early biomarkers for primary hATTR.

Entities:  

Keywords:  Optical Coherence Tomography (OCT); corneal confocal microscopy (CCM); electroretinogram (ERG); hereditary transthyretin amyloidosis (hATTR); ocular biomarkers; personalized medicine; transthyretin (TTR)

Year:  2021        PMID: 34207092     DOI: 10.3390/genes12060927

Source DB:  PubMed          Journal:  Genes (Basel)        ISSN: 2073-4425            Impact factor:   4.096


  3 in total

1.  Ocular Manifestations in a Chinese Pedigree of Familial Amyloidotic Polyneuropathy Carrying the Transthyretin Mutation c.401A>G (p.Tyr134Cys).

Authors:  Xiaonan Zhuang; Zhongcui Sun; Fengjuan Gao; Min Wang; Wenyi Tang; Wei Liu; Keyan Wang; Jihong Wu; Rui Jiang; Gezhi Xu
Journal:  Genes (Basel)       Date:  2022-05-16       Impact factor: 4.141

2.  Muscle MRI as a Useful Biomarker in Hereditary Transthyretin Amyloidosis: A Pilot Study.

Authors:  Guido Primiano; Tommaso Verdolotti; Gabriella D'Apolito; Andrea Di Paolantonio; Valeria Guglielmino; Angela Romano; Gabriele Lucioli; Marco Luigetti; Serenella Servidei
Journal:  Genes (Basel)       Date:  2021-11-11       Impact factor: 4.096

3.  Case Report: Hereditary transthyretin (ATTRv) amyloidosis: The p.G103R mutation of the transthyretin gene in a Han Chinese family is associated with vitreous hemorrhage.

Authors:  Junhui Shen; Hao Yu; Jijian Lin; Li Zhang; Xiaohong Pan; Zhiqing Chen
Journal:  Front Genet       Date:  2022-09-15       Impact factor: 4.772

  3 in total

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