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Updated: Jun 29, 2026

Assessment of Kidney Function in Mouse Models of Glomerular Disease
Published on: June 30, 2018
Genetic and genomic approaches to glomerulosclerosis
1Department of Medicine, School of Medicine, Case Western Reserve University, Cleveland, OH 44109-1998, USA.
Insights
Genetic studies reveal key proteins involved in kidney disease, offering new therapeutic targets for chronic kidney disease (CKD). Research on familial focal segmental glomerulosclerosis advances understanding of podocyte dysfunction in CKD.
Area of Science:
- Nephrology
- Genetics
- Genomics
Background:
- Chronic kidney disease (CKD) is a progressive, costly condition with high morbidity and mortality.
- While modifiable risk factors exist, CKD management remains challenging.
- Genetic factors contribute to CKD susceptibility, despite complex genotype-phenotype correlations.
Purpose of the Study:
- To review recent genetic and genomic discoveries in familial focal segmental glomerulosclerosis (FSGS).
- To explore the role of genetic approaches in understanding CKD pathogenesis.
- To highlight the significance of podocyte dysfunction in glomerular diseases.
Main Methods:
- Linkage analyses of rare familial FSGS cases.
- Genomic and genetic approaches to identify disease-causing genes.
- Characterization of slit diaphragm protein function.
Main Results:
- Discovery of key slit diaphragm proteins through genetic studies.
- Established the critical role of podocyte dysfunction in glomerular diseases.
- Provided insights into the pathogenesis of FSGS and other CKD causes.
Conclusions:
- Genetic and genomic studies are crucial for understanding CKD mechanisms.
- Podocyte dysfunction is a central factor in various forms of CKD.
- These findings pave the way for identifying novel therapeutic targets for CKD.
Abstract:
Chronic kidney disease (CKD) is common, progressive and expensive to manage. Although modifiable risk factors can be treated and outcomes improved, CKD remains a chronic disease with excessive morbidity and mortality. The completion of the human genome sequence and the advent of methodologies to define gene function provide new opportunities to manage and treat patients with CKD and other chronic diseases. Despite the lack of clear correspondence between genotype and phenotype and an obvious Mendelian inheritance pattern, CKD susceptibility has a genetic basis. In this review, we focus on recent studies of familial focal segmental glomerulosclerosis and the discoveries that have resulted from both genetic and genomic approaches used to understand its pathogenesis. Key slit diaphragm proteins were discovered using linkage analyses of these rare causes of glomerulosclerosis and subsequent work has characterized slit diaphragm function in health and disease. Podocyte dysfunction is now recognized as a key contributor to the functional and histologic derangements that characterize glomerular dysfunction in many common causes of CKD. In aggregate, these studies provide a paradigm for approaches to better define mechanisms of CKD and to identify novel therapeutic targets.
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