Literature DB >> 35278126

Spectrum of NPHS1 and NPHS2 variants in egyptian children with focal segmental glomerular sclerosis: identification of six novel variants and founder effect.

Manal M Thomas1, Heba Mostafa Ahmed2, Sara H El-Dessouky3, Abeer Ramadan4, Osama Ezzat Botrous2, Mohamed S Abdel-Hamid5.   

Abstract

The aim of this study is to screen for variants in NPHS1 and NPHS2, in a cohort of Egyptian children with steroid-resistant nephrotic syndrome (SRNS)/focal segmental glomerulosclerosis (FSGS) and compare the prevalence of such variants among other ethnic groups. The study included 25 patients: 21 children diagnosed clinically as steroid-resistant nephrotic syndrome and confirmed as FSGS by renal biopsy and four patients diagnosed as congenital nephrotic syndrome with FSGS. Mutational analysis revealed nine NPHS2 and NPHS1 variants in 13/25 patients with a pathogenic variant detection rate of 52%. NPHS2 variants were found in 8 patients (32%) while five patients from four unrelated families (20%) harbored variants in NPHS1 gene. Six variants were not described before including a likely founder NPHS2 variant in our population, c.596dupA (p.Asn199LysfsTer14). In conclusion, we reported the largest series of patients with SRNS/FSGS from Egypt and identified many novel NPHS1 and NPHS2 variants expanding their mutational spectrum. Further studies on a larger number of patients could provide new insights into the pathogenic mechanisms of SRNS/FSGS which might help in patient's management and prognosis.
© 2022. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.

Entities:  

Keywords:  Egyptian patients; FSGS; Founder effect; NPHS1; NPHS2; Novel variants; SRNS

Mesh:

Substances:

Year:  2022        PMID: 35278126     DOI: 10.1007/s00438-022-01877-3

Source DB:  PubMed          Journal:  Mol Genet Genomics        ISSN: 1617-4623            Impact factor:   3.291


  45 in total

1.  NPHS2, encoding the glomerular protein podocin, is mutated in autosomal recessive steroid-resistant nephrotic syndrome.

Authors:  N Boute; O Gribouval; S Roselli; F Benessy; H Lee; A Fuchshuber; K Dahan; M C Gubler; P Niaudet; C Antignac
Journal:  Nat Genet       Date:  2000-04       Impact factor: 38.330

2.  NPHS2 gene in steroid-resistant nephrotic syndrome: prevalence, clinical course, and mutational spectrum in South-West Iranian children.

Authors:  Mitra Basiratnia; Majid Yavarian; Simin Torabinezhad; Asma Erjaee
Journal:  Iran J Kidney Dis       Date:  2013-09       Impact factor: 0.892

3.  NPHS2 gene mutations in azerbaijani children with steroid-resistant nephrotic syndrome.

Authors:  Rauf Baylarov; Ozgur Senol; Merve Atan; Afig Berdeli
Journal:  Saudi J Kidney Dis Transpl       Date:  2020 Jan-Feb

4.  Genomic and clinical profiling of a national nephrotic syndrome cohort advocates a precision medicine approach to disease management.

Authors:  Agnieszka Bierzynska; Hugh J McCarthy; Katrina Soderquest; Ethan S Sen; Elizabeth Colby; Wen Y Ding; Marwa M Nabhan; Larissa Kerecuk; Shivram Hegde; David Hughes; Stephen Marks; Sally Feather; Caroline Jones; Nicholas J A Webb; Milos Ognjanovic; Martin Christian; Rodney D Gilbert; Manish D Sinha; Graham M Lord; Michael Simpson; Ania B Koziell; Gavin I Welsh; Moin A Saleem
Journal:  Kidney Int       Date:  2017-01-20       Impact factor: 10.612

5.  Immunosuppression and renal outcome in congenital and pediatric steroid-resistant nephrotic syndrome.

Authors:  Anja K Büscher; Birgitta Kranz; Rainer Büscher; Friedhelm Hildebrandt; Bernd Dworniczak; Petra Pennekamp; Eberhard Kuwertz-Bröking; Anne-Margret Wingen; Ulrike John; Markus Kemper; Leo Monnens; Peter F Hoyer; Stefanie Weber; Martin Konrad
Journal:  Clin J Am Soc Nephrol       Date:  2010-08-26       Impact factor: 8.237

Review 6.  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

7.  Ethnic Differences in Incidence and Outcomes of Childhood Nephrotic Syndrome.

Authors:  Tonny H M Banh; Neesha Hussain-Shamsy; Viral Patel; Jovanka Vasilevska-Ristovska; Karlota Borges; Cathryn Sibbald; Deborah Lipszyc; Josefina Brooke; Denis Geary; Valerie Langlois; Michele Reddon; Rachel Pearl; Leo Levin; Monica Piekut; Christoph P B Licht; Seetha Radhakrishnan; Kimberly Aitken-Menezes; Elizabeth Harvey; Diane Hebert; Tino D Piscione; Rulan S Parekh
Journal:  Clin J Am Soc Nephrol       Date:  2016-07-21       Impact factor: 8.237

8.  A spectrum of novel NPHS1 and NPHS2 gene mutations in pediatric nephrotic syndrome patients from Pakistan.

Authors:  Aiysha Abid; Shagufta Khaliq; Saba Shahid; Ali Lanewala; Mohammad Mubarak; Seema Hashmi; Javed Kazi; Tahir Masood; Farkhanda Hafeez; Syed Ali Anwar Naqvi; Syed Adeebul Hasan Rizvi; Syed Qasim Mehdi
Journal:  Gene       Date:  2012-04-28       Impact factor: 3.688

9.  Targeted next-generation sequencing in steroid-resistant nephrotic syndrome: mutations in multiple glomerular genes may influence disease severity.

Authors:  Gemma Bullich; Daniel Trujillano; Sheila Santín; Stephan Ossowski; Santiago Mendizábal; Gloria Fraga; Álvaro Madrid; Gema Ariceta; José Ballarín; Roser Torra; Xavier Estivill; Elisabet Ars
Journal:  Eur J Hum Genet       Date:  2014-11-19       Impact factor: 4.246

Review 10.  NPHS2 mutations in steroid-resistant nephrotic syndrome: a mutation update and the associated phenotypic spectrum.

Authors:  Karim Bouchireb; Olivia Boyer; Olivier Gribouval; Fabien Nevo; Evelyne Huynh-Cong; Vincent Morinière; Raphaëlle Campait; Elisabet Ars; Damien Brackman; Jacques Dantal; Philippe Eckart; Maddalena Gigante; Beata S Lipska; Aurélia Liutkus; André Megarbane; Nabil Mohsin; Fatih Ozaltin; Moin A Saleem; Franz Schaefer; Kenza Soulami; Roser Torra; Nicolas Garcelon; Géraldine Mollet; Karin Dahan; Corinne Antignac
Journal:  Hum Mutat       Date:  2013-12-09       Impact factor: 4.878

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