Evidence map›Paper›PMID 39566961›Full record

ReviewEndocrine journal2025

A parent and child with Liddle syndrome diagnosed correctly with the child as the proband: a case report with review of literature.

Minako Tokunaga, Yuko Seki, Tatsushi Horiguchi, Kiwako Miura, Haruna Kakimoto, Satoshi Morita, Michiyo Mizota, Koshi Kusumoto, Takayasu Mori, Eisei Sohara and 2 more

Abstract readCase ReportsReview
In one paragraph

Review in Endocrine journal, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed, 1 pooled it
–field-weighted citation impact
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, 1 synthesis or guideline pooled it.

  1. Pooled it
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

12 authors.

Minako TokunagaDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.
Yuko SekiDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.
Tatsushi HoriguchiDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.
Kiwako MiuraDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.
Haruna KakimotoDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.ORCID http://orcid.org/0000-0003-2600-5910
Satoshi MoritaDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.ORCID http://orcid.org/0000-0002-1014-1646
Michiyo MizotaDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.
Koshi KusumotoDepartment of Diabetes and Endocrinology, Kagoshima University Hospital, Kagoshima 890-8520, Japan.
Takayasu MoriDepartment of Nephrology, Institute of Science Tokyo, Tokyo 113-8519, Japan.ORCID http://orcid.org/0000-0002-9308-7787
Eisei SoharaDepartment of Nephrology, Institute of Science Tokyo, Tokyo 113-8519, Japan.
Shinichi UchidaDepartment of Nephrology, Institute of Science Tokyo, Tokyo 113-8519, Japan.
Yasuhiro OkamotoDepartment of Pediatrics, Kagoshima University Hospital, Kagoshima 890-8520, Japan.ORCID http://orcid.org/0000-0003-4412-6441

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Liddle syndrome (LS) is an autosomal dominant genetic disorder characterized by early onset hypertension, hypokalemia, and low plasma aldosterone or renin concentration. It is caused by mutations in subunits of the epithelial sodium channel (ENaC). The clinical phenotypes of LS are variable and nonspecific, making it prone to both misdiagnosis and missed diagnosis. Genetic analysis is necessary to confirm the diagnosis of LS. Herein, we report the case of a 42-year-old male with LS and a 30-year history of hypertension. He was being treated for possible primary aldosteronism (PA) over the preceding 7 years; however, his hypertension was poorly controlled despite intensive combination therapy. His 13-year-old son served as a proband for a diagnosis of LS, as he had hypertension, hypokalemia, and a significant family history of hypertension. Genetic testing revealed a heterozygous pathological variant in the SCNN1B gene. This led to a diagnosis of LS, as the father was found to harbor the same mutation. Both were treated with ENaC inhibitors and a salt-restricted diet, which improved their symptoms markedly. The son's genetic diagnosis facilitated the subsequent proper diagnosis and treatment of his father. LS causes early onset hypertension; hence, its early diagnosis and treatment can prevent complications. Hereditary hypertension should be considered in cases of early onset hypertension with a significant family history. Patients diagnosed with PA using outdated criteria may have concomitant LS and require careful evaluation of biochemical and endocrine tests according to the current criteria.

Indexed as

HypertensionLiddle SyndromeAdolescentAdultDiagnostic ErrorsEpithelial Sodium ChannelsHumansHypokalemiaMaleMutationEpithelial Sodium ChannelsSCNN1B protein, humanEarly onset hypertensionHypokalemiaLiddle syndromePrimary aldosteronismSCNN1B mutation

Identifiers

PMID39566961
PMCPMC11913552

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Registered trials

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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.