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Bilateral Symmetry of Visual Function Loss in Cone–Rod Dystrophies

Purpose: To investigate bilateral symmetry of visual impairment in cone–rod dystrophy (CRD) patients and understand the feasibility of clinical trial designs treating one eye and using the untreated eye as an internal control.

Methods: This was a retrospective study of visual function loss measures in 436 CRD patients followed at the Ophthalmology Department of the Catholic University in Rome. Clinical measures considered were best-corrected visual acuity, focal macular cone electroretinogram (fERG), and Ganzfeld cone-mediated and rod-mediated electroretinograms. Interocular agreement in each of these clinical indexes was assessed by t- and Wilcoxon tests for paired samples, structural (Deming) regression analysis, and intraclass correlation. Baseline and follow-up measures were analyzed. A separate analysis was performed on the subset of 61 CRD patients carrying likely disease-causing mutations in the ABCA4 gene.

Results: Statistical tests show a very high degree of bilateral symmetry in the extent and progression of visual impairment in the fellow eyes of CRD patients.

Conclusions: These data contribute to a better understanding of CRDs and support the feasibility of clinical trial designs involving unilateral eye treatment with the use of fellow eye as internal control.

 


Invest Ophthalmol Vis Sci, 2016

Autori esterni: Lucia Galli-Resta (Istituto di Neurofisiologia CNR), Benedetto Falsini (Istituto di Oftalmologia, Università Cattolica del S. Cuore), Giuseppe Rossi (Istituto di Fisiologia Clinica CNR, Pisa, Italy), Marco Piccardi (Institute of Ophthalmology, Policlinico Gemelli, Catholic University, Rome, Italy), Lucia Ziccardi (Bietti Foundation, Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Rome, Italy), Antonello Fadda (Tecnologie e Salute, Istituto Superiore di Sanità, Rome, Italy), Angelo Minnella (Institute of Ophthalmology, Policlinico Gemelli, Catholic University, Rome, Italy), Dario Marangoni (Institute of Ophthalmology, Policlinico Gemelli, Catholic University, Rome, Italy), Giorgio Placidi (Institute of Ophthalmology, Policlinico Gemelli, Catholic University, Rome, Italy), Francesca Campagna (Institute of Ophthalmology, Policlinico Gemelli, Catholic University, Rome, Italy), Edoardo Abed (Institute of Ophthalmology, Policlinico Gemelli, Catholic University, Rome, Italy), Matteo Bertelli (MAGI Laboratory for Medical Genetics, Rovereto, Italy), Monia Zuntini (MAGI Laboratory for Medical Genetics, Rovereto, Italy)
Autori IIT:

Tipo: Contributo in rivista ISI
Area di disciplina: Mathematics

File: i1552-5783-57-8-3759.pdf

Attività: Biologia computazionale