Neuro-Ophthalmological Findings in Friedreich’s Ataxia

Friedreich ataxia (FRDA) is a progressive neurodegenerative disease caused by a severe autosomal recessive genetic disorder of the central nervous (CNS) and peripheral nervous system (PNS), affecting children and young adults. Its onset is before 25 years of age, with mean ages of onset and death be...

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Detalles Bibliográficos
Autores: Rojas Lozano, María Del Pilar, Hoz Montañana, María Rosa De, Cadena Santoyo, Manuel, García Martín, Elena Salobrar, Fernández Albarral, José, López Cuenca, Inés, Elvira Hurtado, Lorena, Urcelay Segura, José Luis, Salazar Corral, Juan José, Ramírez Sebastián, José Manuel, Ramírez Sebastián, Ana Isabel
Tipo de recurso: artículo
Fecha de publicación:2021
País:España
Institución:Universidad Complutense de Madrid (UCM)
Repositorio:Docta Complutense
Idioma:inglés
OAI Identifier:oai:docta.ucm.es:20.500.14352/4438
Acceso en línea:https://hdl.handle.net/20.500.14352/4438
Access Level:acceso abierto
Palabra clave:617.731-003.8
616.8‑009.26
616.832.61
Friedreich ataxia
FRDA
Neurodegeneration
Neurological disability
Eye
Retina
Neurociencias (Medicina)
Oftalmología
2490 Neurociencias
3201.09 Oftalmología
Descripción
Sumario:Friedreich ataxia (FRDA) is a progressive neurodegenerative disease caused by a severe autosomal recessive genetic disorder of the central nervous (CNS) and peripheral nervous system (PNS), affecting children and young adults. Its onset is before 25 years of age, with mean ages of onset and death between 11 and 38 years, respectively. The incidence is 1 in 30,000–50,000 persons. It is caused, in 97% of cases, by a homozygous guanine-adenine-adenine (GAA) trinucleotide mutation in the first intron of the frataxin (FXN) gene on chromosome 9 (9q13–q1.1). The mutation of this gene causes a deficiency of frataxin, which induces an altered inflow of iron into the mitochondria, increasing the nervous system’s vulnerability to oxidative stress. The main clinical signs include spinocerebellar ataxia with sensory loss and disappearance of deep tendon reflexes, cerebellar dysarthria, cardiomyopathy, and scoliosis. Diabetes, hearing loss, and pes cavus may also occur, and although most patients with FRDA do not present with symptomatic visual impairment, 73% present with clinical neuro-ophthalmological alterations such as optic atrophy and altered eye movement, among others. This review provides a brief overview of the main aspects of FRDA and then focuses on the ocular involvement of this pathology and the possible use of retinal biomarkers.