Autosomal Dominant Optic Atrophy plus syndrome (ADOA, OMIM #125250) is a mitochondrial optic neuropathy characterized by progressive degeneration of retinal ganglion cells (RGCs), leading to worsening visual impairment. The disease is caused by pathogenic variants in the Optic Atrophy 1 (OPA1) gene, a member of the guanosine triphosphatase (GTPase) family that plays a central role in mitochondrial fusion and fission, mitophagy regulation, and mitochondrial DNA (mtDNA) maintenance. To model this disorder, we generated and characterized a human induced pluripotent stem cell (hiPSC) line from primary fibroblasts obtained from a patient affected by ADOA syndrome.

Giada Giovenale, A., Ferrone, I., Tomaselli, S., Turco, E., Mazzoni, M., Torres, B., et al. (2026). Generation and characterization of the hiPSC line CSSi023-A (16154) from a patient with ADOA caused by an OPA1 variant. STEM CELL RESEARCH, 94(August 2026) [10.1016/j.scr.2026.104022].

Generation and characterization of the hiPSC line CSSi023-A (16154) from a patient with ADOA caused by an OPA1 variant

Vulcano E.;Ferrari D.;
2026

Abstract

Autosomal Dominant Optic Atrophy plus syndrome (ADOA, OMIM #125250) is a mitochondrial optic neuropathy characterized by progressive degeneration of retinal ganglion cells (RGCs), leading to worsening visual impairment. The disease is caused by pathogenic variants in the Optic Atrophy 1 (OPA1) gene, a member of the guanosine triphosphatase (GTPase) family that plays a central role in mitochondrial fusion and fission, mitophagy regulation, and mitochondrial DNA (mtDNA) maintenance. To model this disorder, we generated and characterized a human induced pluripotent stem cell (hiPSC) line from primary fibroblasts obtained from a patient affected by ADOA syndrome.
Articolo in rivista - Articolo scientifico
iPSCs
English
1-giu-2026
2026
94
August 2026
104022
open
Giada Giovenale, A., Ferrone, I., Tomaselli, S., Turco, E., Mazzoni, M., Torres, B., et al. (2026). Generation and characterization of the hiPSC line CSSi023-A (16154) from a patient with ADOA caused by an OPA1 variant. STEM CELL RESEARCH, 94(August 2026) [10.1016/j.scr.2026.104022].
File in questo prodotto:
File Dimensione Formato  
Giovenale et al-2026-Stem Cell Research-VoR.pdf

accesso aperto

Tipologia di allegato: Publisher’s Version (Version of Record, VoR)
Licenza: Creative Commons
Dimensione 4.25 MB
Formato Adobe PDF
4.25 MB Adobe PDF Visualizza/Apri

I documenti in IRIS sono protetti da copyright e tutti i diritti sono riservati, salvo diversa indicazione.

Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/10281/617721
Citazioni
  • Scopus 0
  • ???jsp.display-item.citation.isi??? 0
Social impact