Symbol
| GUCY2D
| contributors: shn/npt/pgu - updated : 21-05-2015
|
HGNC name
| guanylate cyclase 2D, membrane (retina-specific)
|
HGNC id
| 4689
|
Other morbid association(s)
|
Type | Gene Modification | Chromosome rearrangement | Protein expression | Protein Function
|
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constitutional
| somatic mutation
|  
|  
| loss of function
|
complete lose of function of RetGC1 (GUCY2D) in LCA | constitutional
| somatic mutation
|  
| --over
| gain of function
|
CORD6 mutations result in failure to inactivate cyclase activity at high physiological Ca2+ concentrations. RetGC1 is constituvely activated by its activators GCAP1 and GCAP2 | constitutional
| germinal mutation
|  
|  
|  
|
in LCA2 (acting as a gene modifier) | |
Variant & Polymorphism
|
| |
Candidate gene
Marker
Therapy target
|
System | Type | Disorder | Pubmed |
neurosensorial | visual | degenerative | |
delivery of AAV5 vectors containing murine guanylate cyclase-1 (GC1) cDNA is capable of restoring cone-mediated function and visual behavior and preserving cone photoreceptors in the GC1 knockout (GC1KO) mouse for 3 months | neurosensorial | visual | degenerative | |
AAV8(Y733F) is the most efficient vector for Cone function restauratioon in guanylate cyclase-1 knockout (GC1KO) mouse which is preserved for at least 11 months after treatment |
| | | |
| Gucy2d KO mice develop a cone dystrophy. | |
A 22-kb deletion in the chicken GUCY2D orthologue, GUCY1B leads to a retinal dystrophy in the central retina and proceeds progressively towards the periphery |
|
blindness associated with loss of function of retGC1 in the GUCY1*B avian model of LCA1 can be reversed using viral vector-mediated gene transfer ( |
|
Gucy2f knockdown Zebrafish display significantly lower vision and loss and shortening of cone and rod outer segments leading to early visual dysfunction and photoreceptor layer dystrophy ( |