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Retinal disorders

Gene: RP1L1

Green List (high evidence)

RP1L1 (RP1 like 1)
EnsemblGeneIds (GRCh38): ENSG00000183638
EnsemblGeneIds (GRCh37): ENSG00000183638
OMIM: 608581, Gene2Phenotype
RP1L1 is in 1 panel

6 reviews

Chris Campbell (Manchester University NHS Foundation Trust)

Green List (high evidence)

Exon 4 of RP1L1 is repetitive, highly polymorphic, difficult to sequence and multiple stop or frameshift variants from midway through the exon are observed in the GnomAD datset with high MAF/homozygotes occurences. Bialleleic late truncating variants are unlikely to represent a disease mechanism and are not considered to be clinically useful for reporting.
Created: 18 Jul 2025, 3:27 p.m. | Last Modified: 18 Jul 2025, 3:27 p.m.
Panel Version: 8.9

Variants in this GENE are reported as part of current diagnostic practice

Eleanor Williams (Genomics England Curator)

The mode of inheritance of this gene has been updated to 'BOTH monoallelic and biallelic, autosomal or pseudoautosomal' following NHS Genomic Medicine Service approval.
Created: 26 Sep 2024, 3:50 p.m. | Last Modified: 26 Sep 2024, 3:50 p.m.
Panel Version: 6.7

Mode of inheritance
BOTH monoallelic and biallelic, autosomal or pseudoautosomal

Arina Puzriakova (Genomics England Curator)

Comment on mode of inheritance: This gene is associated with occult macular dystrophy (monoallelic variants) and retinitis pigmentosa (biallelic variants) (https://omim.org/entry/608581) with sufficient cases reported for each phenotype. Currently the monoallelic phenotype is not represented on any GMS panels.

Following curation and consultation with the Genomics England clinical team, there was agreement that macular dystrophy is part of the phenotypic target for this panel. Based on previous reviews, it is not clear why the monoallelic MOI was overwritten and therefore this gene will be flagged for specialist review to determine whether the MOI should stay as 'biallelic' or be updated to 'both mono- and biallelic'.
Created: 10 Apr 2024, 2:59 p.m. | Last Modified: 10 Apr 2024, 2:59 p.m.
Panel Version: 4.83

Gavin Arno (UCL Institute of Ophthalmology/Moorfields Eye Hospital)

I don't know

occult MD associated with hotspots, arRP associated with biallelic final exon truncations
Created: 30 Aug 2019, 2:12 p.m. | Last Modified: 30 Aug 2019, 2:12 p.m.
Panel Version: 1.159

Mode of inheritance
BIALLELIC, autosomal or pseudoautosomal

Ivone Leong (Genomics England Curator)

Comment on mode of inheritance: Changed from monoallelic to biallelic as monoallelic is associated with Occult macular dystrophy and biallelic is associated with retinitis pigmentosa.
Created: 29 Aug 2019, 3:46 p.m. | Last Modified: 29 Aug 2019, 3:46 p.m.
Panel Version: 1.157

Ellen McDonagh (Genomics England Curator)

This gene is on the Manchester Genetic Retinal Degeneration Conditions panel (covers known genes for isolated progessive retinal degeneration, Leber congenital amaurosis, macular dystrophy, achromatopsia, congenital stationary night blindness as well as the two most common causes of syndromic blindess Usher and Bardet-Biedl syndromes and additional syndromes including Joubert, Senior-Loken, and Cohen syndrome.
Created: 2 Jun 2016, 8:05 a.m.

Details

Mode of Inheritance
BOTH monoallelic and biallelic, autosomal or pseudoautosomal
Sources
  • NHS GMS
  • Expert Review Green
Phenotypes
  • Occult macular dystrophy, OMIM:613587 (AD)
  • Retinitis pigmentosa 88, OMIM:618826 (AR)
OMIM
608581
Clinvar variants
Variants in RP1L1
Penetrance
Complete
Publications
Panels with this gene

History Filter Activity

19 Nov 2024, Gel status: 3

Removed Tag, Removed Tag

Achchuthan Shanmugasundram (Genomics England Curator)

Tag Q2_24_MOI was removed from gene: RP1L1. Tag Q2_24_expert_review was removed from gene: RP1L1.

26 Sep 2024, Gel status: 3

Set mode of inheritance

Sarah Leigh (Genomics England Curator)

Mode of inheritance for gene RP1L1 was changed from BIALLELIC, autosomal or pseudoautosomal to BOTH monoallelic and biallelic, autosomal or pseudoautosomal

10 Apr 2024, Gel status: 3

Set mode of inheritance

Arina Puzriakova (Genomics England Curator)

Mode of inheritance for gene: RP1L1 was changed from BIALLELIC, autosomal or pseudoautosomal to BIALLELIC, autosomal or pseudoautosomal

10 Apr 2024, Gel status: 3

Added Tag, Added Tag

Arina Puzriakova (Genomics England Curator)

Tag Q2_24_MOI tag was added to gene: RP1L1. Tag Q2_24_expert_review tag was added to gene: RP1L1.

7 Nov 2023, Gel status: 3

Set Phenotypes

Arina Puzriakova (Genomics England Curator)

Phenotypes for gene: RP1L1 were changed from Occult Macular Dystrophy; Occult macular dystrophy, 613587 to Occult macular dystrophy, OMIM:613587 (AD); Retinitis pigmentosa 88, OMIM:618826 (AR)

29 Aug 2019, Gel status: 3

Set mode of inheritance

Ivone Leong (Genomics England Curator)

Mode of inheritance for gene: RP1L1 was changed from MONOALLELIC, autosomal or pseudoautosomal, imprinted status unknown to BIALLELIC, autosomal or pseudoautosomal

3 Apr 2019, Gel status: 3

Added New Source, Status Update

Ivone Leong (Genomics England Curator)

Source NHS GMS was added to RP1L1. Rating Changed from Green List (high evidence) to Green List (high evidence)

1 Apr 2019, Gel status: 3

Set publications

Ivone Leong (Genomics England Curator)

Publications for gene: RP1L1 were set to 15563508; 17652713; 15090652

1 Apr 2019, Gel status: 3

Set publications

Ivone Leong (Genomics England Curator)

Publications for gene: RP1L1 were set to

22 Mar 2016, Gel status: 4

Gene classified by Genomics England curator

Ellen McDonagh (Genomics England Curator)

This gene has been classified as Green List (High Evidence).

9 Mar 2016, Gel status: 4

Set Mode of Inheritance

Ellen McDonagh (Genomics England Curator)

Model of inheritance for gene RP1L1 was set to MONOALLELIC, autosomal or pseudoautosomal, imprinted status unknown

9 Mar 2016, Gel status: 0

Created

Ellen McDonagh (Genomics England Curator)

RP1L1 was created by ellenmcdonagh

9 Mar 2016, Gel status: 4

Added New Source

Ellen McDonagh (Genomics England Curator)

RP1L1 was added to Posterior segment abnormalitiespanel. Sources: Expert Review Green