Evidence map›Paper›PMID 29513881›Full record

ArticleBrazilian journal of medical and biological research = Revista brasileira de pesquisas medicas e biologica2018

Identification of a novel UMOD mutation (c.163G>A) in a Brazilian family with autosomal dominant tubulointerstitial kidney disease.

L B Lopes, C C Abreu, C F Souza, L E R Guimaraes, A A Silva, F Aguiar-Alves, K O Kidd, S Kmoch, A J Bleyer, J R Almeida

Open access · goldAbstract readCase Reports
In one paragraph

Article in Brazilian journal of medical and biological research = Revista brasileira de pesquisas medicas e biologica, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.2field-weighted citation impact, top 46% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed, 3 citations in OpenAlex.

  1. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

10 authors at 3 institutions in 3 countries.

L B LopesLaboratório Multiusuário de Apoio è Pesquisa em Nefrologia e Ciências Médicas (LAMAP), Departamento de Patologia, Faculdade de Medicina, Universidade Federal Fluminense, Niterói, RJ, Brasil.
C C AbreuLaboratório Multiusuário de Apoio è Pesquisa em Nefrologia e Ciências Médicas (LAMAP), Departamento de Medicina Clínica, Faculdade de Medicina, Universidade Federal Fluminense, Niterói, RJ, Brasil.
C F SouzaLaboratório Multiusuário de Apoio è Pesquisa em Nefrologia e Ciências Médicas (LAMAP), Departamento de Medicina Clínica, Faculdade de Medicina, Universidade Federal Fluminense, Niterói, RJ, Brasil.
L E R GuimaraesLaboratório Multiusuário de Apoio è Pesquisa em Nefrologia e Ciências Médicas (LAMAP), Departamento de Medicina Clínica, Faculdade de Medicina, Universidade Federal Fluminense, Niterói, RJ, Brasil.
A A SilvaLaboratório Multiusuário de Apoio è Pesquisa em Nefrologia e Ciências Médicas (LAMAP), Departamento de Patologia, Faculdade de Medicina, Universidade Federal Fluminense, Niterói, RJ, Brasil.
F Aguiar-AlvesPrograma de Pós-Graduação em Patologia, Faculdade de Medicina e Laboratório Rodolpho Albino, Universidade Federal Fluminense, Niterói, RJ, Brasil.
K O KiddSection on Nephrology, Wake Forest School of Medicine, Winston-Salem, NC, USA.
S KmochInstitute for Inherited Metabolic Disorders, and First Faculty of Medicine, Charles University in Prague, Prague, Czech Republic.
A J BleyerSection on Nephrology, Wake Forest School of Medicine, Winston-Salem, NC, USA.
J R AlmeidaLaboratório Multiusuário de Apoio è Pesquisa em Nefrologia e Ciências Médicas (LAMAP), Departamento de Medicina Clínica, Faculdade de Medicina, Universidade Federal Fluminense, Niterói, RJ, Brasil.
Universidade Federal Fluminense · BRWake Forest University · USCharles University · CZ

Funding

Genetic Variation in Age of Onset of Kidney Failure in Uromodulin Kidney DiseaseR21DK106584 · NIDDK · WAKE FOREST UNIVERSITY HEALTH SCIENCES · PI BLEYER, ANTHONY J · 2015 to 2016
$426k
NIDDK NIH HHS R21 DK106584
6 · The paper itself

Abstract

Autosomal dominant tubulointerstitial kidney disease (ADTKD) is characterized by autosomal dominant inheritance, progressive chronic kidney disease, and a bland urinary sediment. ADTKD is most commonly caused by mutations in the UMOD gene encoding uromodulin (ADTKD-UMOD). We herein report the first confirmed case of a multi-generational Brazilian family with ADTKD-UMOD, caused by a novel heterozygous mutation (c.163G>A, GGC→AGC, p.Gly55Ser) in the UMOD gene. Of 41 family members, 22 underwent genetic analysis, with 11 individuals found to have this mutation. Three affected individuals underwent hemodialysis, one peritoneal dialysis, and one patient received a kidney transplant from a family member later found to be genetically affected. Several younger individuals affected with the mutation were also identified. Clinical characteristics included a bland urinary sediment in all tested individuals and a kidney biopsy in one individual showing tubulointerstitial fibrosis. Unlike most other reported families with ADTKD-UMOD, neither gout nor hyperuricemia was found in affected individuals. In summary, we report a novel UMOD mutation in a Brazilian family with 11 affected members, and we discuss the importance of performing genetic testing in families with inherited kidney disease of unknown cause.

Indexed as

BiopsyFemaleGenotypeHumansMiddle AgedMutationPedigreePolycystic Kidney, Autosomal DominantUromodulinUMOD protein, humanUromodulin

Identifiers

PMID29513881
PMCPMC5912098
OpenAlexW2789314589

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.