| Literature DB >> 25060053 |
Sabrina Giglio1, Aldesia Provenzano2, Benedetta Mazzinghi3, Francesca Becherucci4, Laura Giunti3, Giulia Sansavini4, Fiammetta Ravaglia4, Rosa Maria Roperto4, Silvia Farsetti4, Elisa Benetti5, Mario Rotondi6, Luisa Murer5, Elena Lazzeri7, Laura Lasagni7, Marco Materassi4, Paola Romagnani8.
Abstract
In children, sporadic nephrotic syndrome can be related to a genetic cause, but to what extent genetic alterations associate with resistance to immunosuppression is unknown. In this study, we designed a custom array for next-generation sequencing analysis of 19 target genes, reported as possible causes of nephrotic syndrome, in a cohort of 31 children affected by sporadic steroid-resistant nephrotic syndrome and 38 patients who exhibited a similar but steroid-sensitive clinical phenotype. Patients who exhibited extrarenal symptoms, had a familial history of the disease or consanguinity, or had a congenital onset were excluded. We identified a genetic cause in 32.3% of the children with steroid-resistant disease but zero of 38 children with steroid-sensitive disease. Genetic alterations also associated with lack of response to immunosuppressive agents in children with steroid-resistant disease (0% of patients with alterations versus 57.9% of patients without alterations responded to immunosuppressive agents), whereas clinical features, age at onset, and pathologic findings were similar in steroid-resistant patients with and without alterations. These results suggest that heterogeneous genetic alterations in children with sporadic forms of nephrotic syndrome associate with resistance to steroids as well as immunosuppressive treatments. In these patients, a comprehensive screening using such an array may, thus, be useful for genetic counseling and may help clinical decision making in a fast and cost-efficient manner.Entities:
Keywords: genetic variant; high-throughput DNA sequencing; immunosuppression; proteinuria
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Year: 2014 PMID: 25060053 PMCID: PMC4279734 DOI: 10.1681/ASN.2013111155
Source DB: PubMed Journal: J Am Soc Nephrol ISSN: 1046-6673 Impact factor: 10.121