Retinal ciliopathy due to mutation in nephronophthisis gene

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ORPHA:156180
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Overview

Retinal ciliopathy due to mutation in nephronophthisis genes (Orphanet code 156180) refers to a group of inherited retinal dystrophies caused by mutations in genes that are typically associated with nephronophthisis (NPHP), a ciliopathy primarily known for causing cystic kidney disease. These genes encode proteins essential for the structure and function of primary cilia, which play critical roles in cellular signaling in multiple organ systems, including the retina. When mutations in NPHP genes predominantly or exclusively affect the eye, patients develop progressive retinal degeneration without necessarily manifesting the renal features classically associated with nephronophthisis. The retinal involvement typically presents as a rod-cone dystrophy or retinitis pigmentosa-like phenotype, with symptoms including progressive loss of night vision (nyctalopia), constriction of peripheral visual fields, and eventual decline in central visual acuity. Some patients may also develop features of Leber congenital amaurosis or early-onset severe retinal dystrophy. The condition primarily affects the photoreceptor cells of the retina, which are highly dependent on ciliary function for the transport of proteins within the outer segments. Genes implicated include NPHP1, NPHP4, IQCB1 (NPHP5), CEP290 (NPHP6), and others within the nephronophthisis gene family. There is currently no curative treatment for retinal ciliopathy due to NPHP gene mutations. Management is primarily supportive and includes regular ophthalmological monitoring, low-vision aids, and orientation and mobility training. Gene therapy approaches are under active investigation for some forms of retinal ciliopathies, and patients should be monitored for potential renal involvement, as kidney disease may develop later in life even in those initially presenting with isolated retinal disease. Genetic counseling is recommended for affected families.

Inheritance

Autosomal recessive

Passed on when both parents carry the same gene change; often skips generations

Age of Onset

Variable

Can begin at different ages, from infancy through adulthood

Orphanet ↗NORD ↗

Treatments

No FDA-approved treatments are currently listed for Retinal ciliopathy due to mutation in nephronophthisis gene.

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Treatment Centers

8 centers
🏥 NORD

Baylor College of Medicine Rare Disease Center

Baylor College of Medicine

📍 Houston, TX

🏥 NORD

Stanford Medicine Rare Disease Center

Stanford Medicine

📍 Stanford, CA

🔬 UDN

NIH Clinical Center Undiagnosed Diseases Program

National Institutes of Health

📍 Bethesda, MD

🔬 UDN

UCLA UDN Clinical Site

UCLA Health

📍 Los Angeles, CA

🔬 UDN

Baylor College of Medicine UDN Clinical Site

Baylor College of Medicine

📍 Houston, TX

🔬 UDN

Harvard/MGH UDN Clinical Site

Massachusetts General Hospital

📍 Boston, MA

🏥 NORD

Mayo Clinic Center for Individualized Medicine

Mayo Clinic

📍 Rochester, MN

👤 Mayo Clinic Center for Individualized Medicine

🏥 NORD

UCLA Rare Disease Day Program

UCLA Health

📍 Los Angeles, CA

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Common questions about Retinal ciliopathy due to mutation in nephronophthisis gene

What is Retinal ciliopathy due to mutation in nephronophthisis gene?

Retinal ciliopathy due to mutation in nephronophthisis genes (Orphanet code 156180) refers to a group of inherited retinal dystrophies caused by mutations in genes that are typically associated with nephronophthisis (NPHP), a ciliopathy primarily known for causing cystic kidney disease. These genes encode proteins essential for the structure and function of primary cilia, which play critical roles in cellular signaling in multiple organ systems, including the retina. When mutations in NPHP genes predominantly or exclusively affect the eye, patients develop progressive retinal degeneration with

How is Retinal ciliopathy due to mutation in nephronophthisis gene inherited?

Retinal ciliopathy due to mutation in nephronophthisis gene follows a autosomal recessive inheritance pattern. Genetic counseling can help families understand recurrence risk and testing options.