Evidence map›Paper›PMID 35327948›Full record

ArticleGenes2022

Rapidly Progressing to ESRD in an Individual with Coexisting ADPKD and Masked Klinefelter and Gitelman Syndromes.

Ramón Peces, Carlos Peces, Rocío Mena, Emilio Cuesta, Fe Amalia García-Santiago, Marta Ossorio, Sara Afonso, Pablo Lapunzina, Julián Nevado

Open access · goldAbstract readCase Reports
In one paragraph

Article in Genes, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
0.4field-weighted citation impact, top 34% 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

0 citing papers in PubMed, 2 citations in OpenAlex.

No citing paper in PubMed yet.

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

9 authors at 2 institutions in 1 country.

Ramón PecesServicio de Nefrología, Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.ORCID 0000-0002-9170-8746
Carlos PecesArea de Tecnología de la Información, SESCAM, 45071 Toledo, Spain.
Rocío MenaInstituto de Genética Médica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.
Emilio CuestaServicio de Radiología, Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.
Fe Amalia García-SantiagoInstituto de Genética Médica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.ORCID 0000-0003-2393-7800
Marta OssorioServicio de Nefrología, Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.
Sara AfonsoServicio de Nefrología, Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.
Pablo LapunzinaInstituto de Genética Médica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.ORCID 0000-0002-6324-4825
Julián NevadoInstituto de Genética Médica y Molecular (INGEMM), Hospital Universitario La Paz, IdiPAZ, Universidad Autónoma, 28046 Madrid, Spain.ORCID 0000-0001-5611-2659
Hospital Universitario La Paz · ESServicio de Salud de Castilla La Mancha · ES

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Autosomal dominant polycystic kidney disease (ADPKD) is the most common monogenetic hereditary renal disease, promoting end-stage renal disease (ESRD). Klinefelter syndrome (KS) is a consequence of an extra copy of the X chromosome in males. Main symptoms in KS include hypogonadism, tall stature, azoospermia, and a risk of cardiovascular diseases, among others. Gitelman syndrome (GS) is an autosomal recessive disorder caused by SLC12A3 variants, and is associated with hypokalemia, hypomagnesemia, hypocalciuria, normal or low blood pressure, and salt loss. The three disorders have distinct and well-delineated clinical, biochemical, and genetic findings. We here report a male patient with ADPKD who developed early chronic renal failure leading to ESRD, presenting with an intracranial aneurysm and infertility. NGS identified two de novo PKD1 variants, one known (likely pathogenic), and a previously unreported variant of uncertain significance, together with two SLC12A3 pathogenic variants. In addition, cytogenetic analysis showed a 47, XXY karyotype. We investigated the putative impact of this rare association by analyzing possible clinical, biochemical, and/or genetic interactions and by comparing the evolution of renal size and function in the proband with three age-matched ADPKD (by variants in PKD1) cohorts. We hypothesize that the coexistence of these three genetic disorders may act as modifiers with possible synergistic actions that could lead, in our patient, to a rapid ADPKD progression.

Indexed as

Gitelman SyndromeKidney Failure, ChronicPolycystic Kidney, Autosomal DominantRenal Insufficiency, ChronicHumansKidneyMaleSolute Carrier Family 12, Member 3SLC12A3 protein, humanSolute Carrier Family 12, Member 3ADPKDapoptosischronic kidney disease progressionfibrosisGitelman syndromeintracranial aneurysmKlinefelter syndrome

Identifiers

PMID35327948
PMCPMC8954516
OpenAlexW4214699094

What OpenQuestion holds

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LicenceCC BY
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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.