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Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India

Pediatric nephrolithiasis (NL) or Kidney stone disease (KSD) is an untethered topic in Asian population. In Western countries, the annual incidence of paediatric NL is around 6–10%. Here, we present data from West Bengal, India, on lower age (LA, 0–20 years) NL and its prevalence for the first time....

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Autores principales: Chatterjee, Arindam, Sarkar, Kunal, Bank, Sarbashri, Ghosh, Sudakshina, Kumar Pal, Dilip, Saraf, Siddharth, Wakle, Dhansagar, Roy, Bidyut, Chakraborty, Santanu, Bankura, Biswabandhu, Chattopadhyay, Debprasad, Das, Madhusudan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9815608/
https://www.ncbi.nlm.nih.gov/pubmed/36619171
http://dx.doi.org/10.3389/fmolb.2022.1049620
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author Chatterjee, Arindam
Sarkar, Kunal
Bank, Sarbashri
Ghosh, Sudakshina
Kumar Pal, Dilip
Saraf, Siddharth
Wakle, Dhansagar
Roy, Bidyut
Chakraborty, Santanu
Bankura, Biswabandhu
Chattopadhyay, Debprasad
Das, Madhusudan
author_facet Chatterjee, Arindam
Sarkar, Kunal
Bank, Sarbashri
Ghosh, Sudakshina
Kumar Pal, Dilip
Saraf, Siddharth
Wakle, Dhansagar
Roy, Bidyut
Chakraborty, Santanu
Bankura, Biswabandhu
Chattopadhyay, Debprasad
Das, Madhusudan
author_sort Chatterjee, Arindam
collection PubMed
description Pediatric nephrolithiasis (NL) or Kidney stone disease (KSD) is an untethered topic in Asian population. In Western countries, the annual incidence of paediatric NL is around 6–10%. Here, we present data from West Bengal, India, on lower age (LA, 0–20 years) NL and its prevalence for the first time. To discover the mutations associated with KSD, twenty-four (18 + 6) rare LA-NL patients were selected for Whole Exome Sequencing (WES) and Sanger sequencing, respectively. It was found that GRHPR c. 494G>A mutation (MZ826703) is predominant in our study cohort. This specific homozygous mutation is functionally studied for the first time directly from human peripheral mononuclear cell (PBMC) samples. Using expression study with biochemical activity and computational analysis we assumed that the mutation is pathogenic with loss of function. Moreover, three genes, AGXT, HOGA1 and GRHPR with Novel variants known to cause hyperoxaluria were found frequently in the study cohort. Our study analyses the genes and variations that cause LA-NL, as well as the molecular function of the GRHPR mutation, which may serve as a clinical marker in the population of West Bengal, Eastern India.
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spelling pubmed-98156082023-01-06 Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India Chatterjee, Arindam Sarkar, Kunal Bank, Sarbashri Ghosh, Sudakshina Kumar Pal, Dilip Saraf, Siddharth Wakle, Dhansagar Roy, Bidyut Chakraborty, Santanu Bankura, Biswabandhu Chattopadhyay, Debprasad Das, Madhusudan Front Mol Biosci Molecular Biosciences Pediatric nephrolithiasis (NL) or Kidney stone disease (KSD) is an untethered topic in Asian population. In Western countries, the annual incidence of paediatric NL is around 6–10%. Here, we present data from West Bengal, India, on lower age (LA, 0–20 years) NL and its prevalence for the first time. To discover the mutations associated with KSD, twenty-four (18 + 6) rare LA-NL patients were selected for Whole Exome Sequencing (WES) and Sanger sequencing, respectively. It was found that GRHPR c. 494G>A mutation (MZ826703) is predominant in our study cohort. This specific homozygous mutation is functionally studied for the first time directly from human peripheral mononuclear cell (PBMC) samples. Using expression study with biochemical activity and computational analysis we assumed that the mutation is pathogenic with loss of function. Moreover, three genes, AGXT, HOGA1 and GRHPR with Novel variants known to cause hyperoxaluria were found frequently in the study cohort. Our study analyses the genes and variations that cause LA-NL, as well as the molecular function of the GRHPR mutation, which may serve as a clinical marker in the population of West Bengal, Eastern India. Frontiers Media S.A. 2022-12-22 /pmc/articles/PMC9815608/ /pubmed/36619171 http://dx.doi.org/10.3389/fmolb.2022.1049620 Text en Copyright © 2022 Chatterjee, Sarkar, Bank, Ghosh, Kumar Pal, Saraf, Wakle, Roy, Chakraborty, Bankura, Chattopadhyay and Das. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Molecular Biosciences
Chatterjee, Arindam
Sarkar, Kunal
Bank, Sarbashri
Ghosh, Sudakshina
Kumar Pal, Dilip
Saraf, Siddharth
Wakle, Dhansagar
Roy, Bidyut
Chakraborty, Santanu
Bankura, Biswabandhu
Chattopadhyay, Debprasad
Das, Madhusudan
Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India
title Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India
title_full Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India
title_fullStr Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India
title_full_unstemmed Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India
title_short Homozygous GRHPR C.494G>A mutation is deleterious that causes early onset of nephrolithiasis in West Bengal, India
title_sort homozygous grhpr c.494g>a mutation is deleterious that causes early onset of nephrolithiasis in west bengal, india
topic Molecular Biosciences
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9815608/
https://www.ncbi.nlm.nih.gov/pubmed/36619171
http://dx.doi.org/10.3389/fmolb.2022.1049620
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