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Spectrum of NPHS1 and NPHS2 variants in egyptian children with focal segmental glomerular sclerosis: identification of six novel variants and founder effect.
Thomas, Manal M; Ahmed, Heba Mostafa; El-Dessouky, Sara H; Ramadan, Abeer; Botrous, Osama Ezzat; Abdel-Hamid, Mohamed S.
Afiliación
  • Thomas MM; Clinical Genetics Department, Human Genetics and Genome Research Institute, National Research Centre, 33rd El Bohouth st, former El Tahrir st, Dokki, Cairo, 12311, Egypt. nula_m@hotmail.com.
  • Ahmed HM; Department of Pediatrics, Faculty of Medicine, Beni-Suef University, Beni-Suef, Egypt.
  • El-Dessouky SH; Prenatal Diagnosis and Fetal Medicine Department, Human Genetics and Genome Research Institute, National Research Centre, Cairo, Egypt.
  • Ramadan A; Molecular Genetics and Enzymology Department, Human Genetics and Genome Research Institute, National Research Centre, Cairo, Egypt.
  • Botrous OE; Department of Pediatrics, Faculty of Medicine, Beni-Suef University, Beni-Suef, Egypt.
  • Abdel-Hamid MS; Medical Molecular Genetics Department, Human Genetics and Genome Research Institute, National Research Centre, 33rd El Bohouth st, former El Tahrir st, Dokki, Cairo, 12311, Egypt. mohamadnrc@hotmail.com.
Mol Genet Genomics ; 297(3): 689-698, 2022 May.
Article en En | MEDLINE | ID: mdl-35278126
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.
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Texto completo: 1 Base de datos: MEDLINE Asunto principal: Glomeruloesclerosis Focal y Segmentaria / Síndrome Nefrótico Tipo de estudio: Diagnostic_studies / Risk_factors_studies Límite: Child / Humans País/Región como asunto: Africa Idioma: En Revista: Mol Genet Genomics Asunto de la revista: BIOLOGIA MOLECULAR / GENETICA Año: 2022 Tipo del documento: Article País de afiliación: Egipto

Texto completo: 1 Base de datos: MEDLINE Asunto principal: Glomeruloesclerosis Focal y Segmentaria / Síndrome Nefrótico Tipo de estudio: Diagnostic_studies / Risk_factors_studies Límite: Child / Humans País/Región como asunto: Africa Idioma: En Revista: Mol Genet Genomics Asunto de la revista: BIOLOGIA MOLECULAR / GENETICA Año: 2022 Tipo del documento: Article País de afiliación: Egipto