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NCT05793515

Mechanisms of Inherited Retinal Dystrophies Using Whole Genome Sequencing and in Vitro and in Vivo Models

Completed Last updated 18 April 2025
What this trial tests

trial testing whole genome sequencing in Inherited Retinal Dystrophy in 120 participants. Completed in 2 November 2024.

Timeline
15 November 2022
Primary endpoint
2 November 2024
2 November 2024

Quick facts

Lead sponsorIstituto Superiore di Sanità
StatusCompleted
Study typeOBSERVATIONAL
Enrollment120
Start date15 November 2022
Primary completion2 November 2024
Estimated completion2 November 2024
Sites1 location across Italy

Drugs / interventions tested

Conditions studied

Sponsor

Istituto Superiore di Sanità — full company profile →

Who can join

Adults 5 to 80, any sex, with Inherited Retinal Dystrophy or Retinitis Pigmentosa. Patients with the condition only — healthy volunteers not accepted.

Sponsor's own description

Inherited retinal dystrophies (IRDs), a large group of heterogeneous and rare disorders, may result in irreversible bilateral visual loss and blindness. Characterizing the genetic bases of IRDs will help to understand the pathogenesis underlying the development of retinal damage. Despite the advances in molecular identification of genes causing disease, unsolved IRDs constitute about 40% of all cases. Goal of this study is to solve missing heritability in IRD using whole genome sequencing (WGS) to identify the genetic causes in clinically well-characterized patients without a molecular diagnosis. The identification of novel genes that have a role in the development or maintenance of retinal function will lead to the development of new therapeutic approaches and will favour a more prompt diagnosis and improvement of patient management.

Publications & conference data

4 peer-reviewed publications reference this trial (live from Europe PMC):

  1. Bi-allelic variants in SNF8 cause a disease spectrum ranging from severe developmental and epileptic encephalopathy to syndromic optic atrophy.
    Brugger M, Lauri A, Zhen Y, Gramegna LL, et al · · 2024 · cited 11× · PMID 38423010 · DOI 10.1016/j.ajhg.2024.02.005
  2. A Novel Heterozygous ARL3 Variant in Non-Syndromic Retinitis Pigmentosa: Clinical and Functional Characterization
    Stellacci E, Ziccardi L, Bruselles A, Dell’Aquila C, et al · · 2026
  3. Biallelic <i>BAIAP3</i> Variants Are Associated with Isolated Retinitis Pigmentosa.
    Cordeddu V, Flex E, Mignini L, Bruselles A, et al · · 2025 · PMID 40943168 · DOI 10.3390/ijms26178244
  4. Whole Genome Sequencing Solves an Atypical Form of Bardet-Biedl Syndrome: Identification of Novel Pathogenic Variants of <i>BBS9</i>.
    Stellacci E, Niceta M, Bruselles A, Straface E, et al · · 2024 · PMID 39125883 · DOI 10.3390/ijms25158313

Verify or expand the search:

Other trials of whole genome sequencing

Trials testing the same drug.

Other recruiting trials for Inherited Retinal Dystrophy

Currently open trials in the same condition.

Other Istituto Superiore di Sanità trials

Trials by the same sponsor.

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Data sources for this page

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