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Whole-genome sequencing of Finnish Type 1 diabetic siblings discordant for kidney disease reveals DNA variants associated with diabetic nephropathy

Whole-genome sequencing of Finnish Type 1 diabetic siblings discordant for kidney disease reveals DNA variants associated with diabetic nephropathy
Whole-genome sequencing of Finnish Type 1 diabetic siblings discordant for kidney disease reveals DNA variants associated with diabetic nephropathy

BACKGROUND: Several genetic susceptibility loci associated with diabetic nephropathy have been documented, but no causative variants implying novel pathogenetic mechanisms have been elucidated.

METHODS: We carried out whole-genome sequencing of a discovery cohort of Finnish siblings with type 1 diabetes who were discordant for the presence (case) or absence (control) of diabetic nephropathy. Controls had diabetes without complications for 15-37 years. We analyzed and annotated variants at genome, gene, and single-nucleotide variant levels. We then replicated the associated variants, genes, and regions in a replication cohort from the Finnish Diabetic Nephropathy study that included 3531 unrelated Finns with type 1 diabetes.

RESULTS: We observed protein-altering variants and an enrichment of variants in regions associated with the presence or absence of diabetic nephropathy. The replication cohort confirmed variants in both regulatory and protein-coding regions. We also observed that diabetic nephropathy-associated variants, when clustered at the gene level, are enriched in a core protein-interaction network representing proteins essential for podocyte function. These genes include protein kinases (protein kinase C isoforms ε and ι) and protein tyrosine kinase 2.

CONCLUSIONS: Our comprehensive analysis of a diabetic nephropathy cohort of siblings with type 1 diabetes who were discordant for kidney disease points to variants and genes that are potentially causative or protective for diabetic nephropathy. This includes variants in two isoforms of the protein kinase C family not previously linked to diabetic nephropathy, adding support to previous hypotheses that the protein kinase C family members play a role in diabetic nephropathy and might be attractive therapeutic targets.

Adolescent, Adult, Animals, Child, Child, Preschool, Diabetes Mellitus, Type 1/complications, Diabetic Nephropathies/genetics, Female, HEK293 Cells, Humans, Male, Polymorphism, Single Nucleotide, Protein Kinase C/physiology, Siblings, Whole Genome Sequencing/methods, Young Adult, Zebrafish
1046-6673
309-323
Guo, Jing
e51fdc35-39a7-4188-9268-5a5d03ad908a
Rackham, Owen J L
8122eb1f-6e9f-4da5-90e1-ce108ccbbcbf
Sandholm, Niina
f3a03b02-ed6a-44fd-9b69-4e9349da2a66
He, Bing
be7a2c34-6ba4-42fc-9351-47fd6e7ed9a9
Österholm, Anne-May
2bb86765-e52c-4009-b18a-7dddb5850f34
Valo, Erkka
5b5add09-a5ad-44fa-9f93-6ccf0500faf8
Harjutsalo, Valma
b276d7fa-e04f-441d-b341-d8b0bc497e56
Forsblom, Carol
1a548f7a-184a-4a92-8fbc-738af0631c66
Toppila, Iiro
77dcda13-789f-4420-a943-29715f6d3638
Parkkonen, Maija
da34dc95-635b-4958-9cef-eb958ed11437
Li, Qibin
5316a954-61ce-49c2-b3f3-8a6ccc6744e6
Zhu, Wenjuan
42be32ad-3ea7-4684-9505-f75ae615adfc
Harmston, Nathan
017e019d-7784-4ed4-a4d9-38880f08df3f
Chothani, Sonia
24850611-01f3-46ae-af99-8c2693e6ca8f
Öhman, Miina K
ce90f81a-5a22-4b96-9c52-b21617877d92
Eng, Eudora
e0bfbee3-d066-4fe9-9eef-6e65015bd51e
Sun, Yang
80cef8d5-7112-44a7-b28c-6fe38013e8e7
Petretto, Enrico
a8a7d254-ea06-4ab3-ba7e-b653349a29f4
Groop, Per-Henrik
71278335-e6e4-41ce-aa65-429f197a3b7a
Tryggvason, Karl
0f60be6a-0f0c-4dbe-8823-baa1971a96b9
Guo, Jing
e51fdc35-39a7-4188-9268-5a5d03ad908a
Rackham, Owen J L
8122eb1f-6e9f-4da5-90e1-ce108ccbbcbf
Sandholm, Niina
f3a03b02-ed6a-44fd-9b69-4e9349da2a66
He, Bing
be7a2c34-6ba4-42fc-9351-47fd6e7ed9a9
Österholm, Anne-May
2bb86765-e52c-4009-b18a-7dddb5850f34
Valo, Erkka
5b5add09-a5ad-44fa-9f93-6ccf0500faf8
Harjutsalo, Valma
b276d7fa-e04f-441d-b341-d8b0bc497e56
Forsblom, Carol
1a548f7a-184a-4a92-8fbc-738af0631c66
Toppila, Iiro
77dcda13-789f-4420-a943-29715f6d3638
Parkkonen, Maija
da34dc95-635b-4958-9cef-eb958ed11437
Li, Qibin
5316a954-61ce-49c2-b3f3-8a6ccc6744e6
Zhu, Wenjuan
42be32ad-3ea7-4684-9505-f75ae615adfc
Harmston, Nathan
017e019d-7784-4ed4-a4d9-38880f08df3f
Chothani, Sonia
24850611-01f3-46ae-af99-8c2693e6ca8f
Öhman, Miina K
ce90f81a-5a22-4b96-9c52-b21617877d92
Eng, Eudora
e0bfbee3-d066-4fe9-9eef-6e65015bd51e
Sun, Yang
80cef8d5-7112-44a7-b28c-6fe38013e8e7
Petretto, Enrico
a8a7d254-ea06-4ab3-ba7e-b653349a29f4
Groop, Per-Henrik
71278335-e6e4-41ce-aa65-429f197a3b7a
Tryggvason, Karl
0f60be6a-0f0c-4dbe-8823-baa1971a96b9

Guo, Jing, Rackham, Owen J L, Sandholm, Niina, He, Bing, Österholm, Anne-May, Valo, Erkka, Harjutsalo, Valma, Forsblom, Carol, Toppila, Iiro, Parkkonen, Maija, Li, Qibin, Zhu, Wenjuan, Harmston, Nathan, Chothani, Sonia, Öhman, Miina K, Eng, Eudora, Sun, Yang, Petretto, Enrico, Groop, Per-Henrik and Tryggvason, Karl (2020) Whole-genome sequencing of Finnish Type 1 diabetic siblings discordant for kidney disease reveals DNA variants associated with diabetic nephropathy. Journal of the American Society of Nephrology, 31 (2), 309-323. (doi:10.1681/ASN.2019030289).

Record type: Article

Abstract

BACKGROUND: Several genetic susceptibility loci associated with diabetic nephropathy have been documented, but no causative variants implying novel pathogenetic mechanisms have been elucidated.

METHODS: We carried out whole-genome sequencing of a discovery cohort of Finnish siblings with type 1 diabetes who were discordant for the presence (case) or absence (control) of diabetic nephropathy. Controls had diabetes without complications for 15-37 years. We analyzed and annotated variants at genome, gene, and single-nucleotide variant levels. We then replicated the associated variants, genes, and regions in a replication cohort from the Finnish Diabetic Nephropathy study that included 3531 unrelated Finns with type 1 diabetes.

RESULTS: We observed protein-altering variants and an enrichment of variants in regions associated with the presence or absence of diabetic nephropathy. The replication cohort confirmed variants in both regulatory and protein-coding regions. We also observed that diabetic nephropathy-associated variants, when clustered at the gene level, are enriched in a core protein-interaction network representing proteins essential for podocyte function. These genes include protein kinases (protein kinase C isoforms ε and ι) and protein tyrosine kinase 2.

CONCLUSIONS: Our comprehensive analysis of a diabetic nephropathy cohort of siblings with type 1 diabetes who were discordant for kidney disease points to variants and genes that are potentially causative or protective for diabetic nephropathy. This includes variants in two isoforms of the protein kinase C family not previously linked to diabetic nephropathy, adding support to previous hypotheses that the protein kinase C family members play a role in diabetic nephropathy and might be attractive therapeutic targets.

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More information

Accepted/In Press date: 19 October 2019
e-pub ahead of print date: 31 January 2020
Published date: February 2020
Additional Information: Copyright © 2020 by the American Society of Nephrology.
Keywords: Adolescent, Adult, Animals, Child, Child, Preschool, Diabetes Mellitus, Type 1/complications, Diabetic Nephropathies/genetics, Female, HEK293 Cells, Humans, Male, Polymorphism, Single Nucleotide, Protein Kinase C/physiology, Siblings, Whole Genome Sequencing/methods, Young Adult, Zebrafish

Identifiers

Local EPrints ID: 447926
URI: http://eprints.soton.ac.uk/id/eprint/447926
ISSN: 1046-6673
PURE UUID: 3f5ccb85-438e-4140-b90d-5a87412925f0
ORCID for Owen J L Rackham: ORCID iD orcid.org/0000-0002-4390-0872

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Date deposited: 26 Mar 2021 17:30
Last modified: 17 Mar 2024 04:03

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Contributors

Author: Jing Guo
Author: Niina Sandholm
Author: Bing He
Author: Anne-May Österholm
Author: Erkka Valo
Author: Valma Harjutsalo
Author: Carol Forsblom
Author: Iiro Toppila
Author: Maija Parkkonen
Author: Qibin Li
Author: Wenjuan Zhu
Author: Nathan Harmston
Author: Sonia Chothani
Author: Miina K Öhman
Author: Eudora Eng
Author: Yang Sun
Author: Enrico Petretto
Author: Per-Henrik Groop
Author: Karl Tryggvason

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