Literature DB >> 34546508

Mutational landscape of TRPC6, WT1, LMX1B, APOL1, PTPRO, PMM2, LAMB2 and WT1 genes associated with Steroid resistant nephrotic syndrome.

Jinal M Thakor1, Glory Parmar1, Kinnari N Mistry2, Sishir Gang3, Dharamshibhai N Rank4, Chaitanya G Joshi5.   

Abstract

BACKGROUND: Nephrotic syndrome appears as a group of symptoms like proteinuria, edema and hyperlipidemia. Identification of monogenic forms revealed the physiology and pathogenesis of the SRNS. METHODS AND
RESULTS: We performed Illumina panel sequencing of seven genes in 90 Indian patients to determine the role of these genetic mutations in nephrotic syndrome prognosis. Samtool was used for variants calling, and SnpEff and Snpsift did variants annotation. Clinical significance and variant classification were performed by the ClinVar database. In SSNS and SRNS patients, we found 0.78% pathogenic and 3.41% likely pathogenic mutations. Pathogenic mutations were found in LAMB2, LMX1B and WT1 genes, while likely pathogenic mutations were found in (6/13) LAMB2, (2/13) LMX1B, (2/13) TRPC6, (2/13) PTPRO and (1/13) PMM2 genes. Approximately 46% likely pathogenic mutations were contributed to the LAMB2 gene in SSNS and SRNS patients. We also detect 30 VUS (variants of uncertain significance), which were found (17/30) pathogenic and (13/30) likely pathogenic by different prediction tools.
CONCLUSIONS: Multigene panels were used for genetic screening of heterogeneous disorders like nephrotic syndrome in the Indian population. We found pathogenic, likely pathogenic and certain VUS, which were responsible for the pathogenesis of the disease. Therefore, mutational analysis of SSNS and SRNS is necessary to avoid adverse effects of corticosteroids, modify the intensity of immunosuppressing agents, and prevent the disease's progression.
© 2021. The Author(s), under exclusive licence to Springer Nature B.V.

Entities:  

Keywords:  Illumina panel sequencing; Indian patients; Steroid resistant nephrotic syndrome (SRNS); Steroid sensitive nephrotic syndrome (SSNS)

Mesh:

Substances:

Year:  2021        PMID: 34546508     DOI: 10.1007/s11033-021-06711-4

Source DB:  PubMed          Journal:  Mol Biol Rep        ISSN: 0301-4851            Impact factor:   2.316


  45 in total

1.  254C>G: a TRPC6 promoter variation associated with enhanced transcription and steroid-resistant nephrotic syndrome in Chinese children.

Authors:  Xin-Yu Kuang; Wen-Yan Huang; Hong Xu; Yu Shi; Xiu-Ling Zhang; Xiao-Ling Niu; Ying Wu; Chuan-Zhong Mei; Xi-Liang Zha; Zhong-Hua Zhao; Zhi-Gang Zhang
Journal:  Pediatr Res       Date:  2013-09-02       Impact factor: 3.756

Review 2.  Focal segmental glomerulosclerosis and chronic kidney disease in pediatric patients.

Authors:  Jeremy Kiffel; Yael Rahimzada; Howard Trachtman
Journal:  Adv Chronic Kidney Dis       Date:  2011-09       Impact factor: 3.620

3.  Prevalence of WT1 mutations in a large cohort of patients with steroid-resistant and steroid-sensitive nephrotic syndrome.

Authors:  Rainer G Ruf; Michael Schultheiss; Anne Lichtenberger; Stephanie M Karle; Isabella Zalewski; Bettina Mucha; Anne Schulze Everding; Thomas Neuhaus; Ludwig Patzer; Christian Plank; Johannes P Haas; Fatih Ozaltin; Anita Imm; Arno Fuchshuber; Aysin Bakkaloglu; Friedhelm Hildebrandt
Journal:  Kidney Int       Date:  2004-08       Impact factor: 10.612

Review 4.  Hereditary nephrotic syndrome: a systematic approach for genetic testing and a review of associated podocyte gene mutations.

Authors:  Geneviève Benoit; Eduardo Machuca; Corinne Antignac
Journal:  Pediatr Nephrol       Date:  2010-03-24       Impact factor: 3.714

5.  Long-term outcome of idiopathic steroid-resistant nephrotic syndrome: a multicenter study.

Authors:  Djalila Mekahli; Aurelia Liutkus; Bruno Ranchin; Anchalee Yu; Lucie Bessenay; Eric Girardin; Rita Van Damme-Lombaerts; Jean-Bernard Palcoux; François Cachat; Marie-Pierre Lavocat; Guylhène Bourdat-Michel; François Nobili; Pierre Cochat
Journal:  Pediatr Nephrol       Date:  2009-03-12       Impact factor: 3.714

6.  Clinical features and long-term outcome of nephrotic syndrome associated with heterozygous NPHS1 and NPHS2 mutations.

Authors:  Gianluca Caridi; Maddalena Gigante; Pietro Ravani; Antonella Trivelli; Giancarlo Barbano; Francesco Scolari; Monica Dagnino; Luisa Murer; Corrado Murtas; Alberto Edefonti; Landino Allegri; Alessandro Amore; Rosanna Coppo; Francesco Emma; Tommaso De Palo; Rosa Penza; Loreto Gesualdo; Gian Marco Ghiggeri
Journal:  Clin J Am Soc Nephrol       Date:  2009-04-30       Impact factor: 8.237

Review 7.  The spectrum of podocytopathies: a unifying view of glomerular diseases.

Authors:  R C Wiggins
Journal:  Kidney Int       Date:  2007-04-04       Impact factor: 10.612

8.  The advent of personal genome sequencing.

Authors:  Radoje Drmanac
Journal:  Genet Med       Date:  2011-03       Impact factor: 8.822

Review 9.  Nephrotic Syndrome: Oedema Formation and Its Treatment With Diuretics.

Authors:  Sanjana Gupta; Ruth J Pepper; Neil Ashman; Stephen B Walsh
Journal:  Front Physiol       Date:  2019-01-15       Impact factor: 4.566

10.  Steroid-resistant idiopathic nephrotic syndrome in children: long-term follow-up and risk factors for end-stage renal disease.

Authors:  Alberto Zagury; Anne Louise de Oliveira; Jose Augusto Araujo Montalvão; Regina Helena Leite Novaes; Vinicius Martins de Sá; Carlos Augusto Pinheiro de Moraes; Marcelo de Sousa Tavares
Journal:  J Bras Nefrol       Date:  2013 Jul-Sep
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