Evidence map›Paper›PMID 36980135›Full record

ArticleChildren (Basel, Switzerland)2023

Frasier Syndrome: A 15-Year-Old Phenotypically Female Adolescent Presenting with Delayed Puberty and Nephropathy.

Qing Shao, Xinglei Xie, Jia Geng, Xiaoling Yang, Wei Li, Yuwei Zhang

Open access · goldFull text readCase Reports
In one paragraph

Article in Children (Basel, Switzerland), 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.5field-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

2 citing papers in PubMed, 3 citations in OpenAlex.

  1. FamilialWorld journal of nephrology · 2026
    Article
  2. UnderstandingCancers · 2023
    Article
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

6 authors at 1 institution in 1 country.

Qing ShaoDepartment of Endocrinology and Metabolism, West China Hospital of Sichuan University, Chengdu 610041, China.
Xinglei XieDepartment of Endocrinology and Metabolism, West China Hospital of Sichuan University, Chengdu 610041, China.
Jia GengInstitute of Rare Diseases, West China Hospital of Sichuan University, Chengdu 610041, China.
Xiaoling YangDepartment of Endocrinology and Metabolism, West China Hospital of Sichuan University, Chengdu 610041, China.
Wei LiOutpatient Department, West China Hospital of Sichuan University, Chengdu 610041, China.
Yuwei ZhangDepartment of Endocrinology and Metabolism, West China Hospital of Sichuan University, Chengdu 610041, China.ORCID 0000-0003-2281-5000
Sichuan University · CN

Funding

Clinical Research Incubation Project of West China Hospital, Sichuan University 2021HXFH009Department of Science and Technology of Sichuan Province 2020YFQ0020Department of Science and Technology of Sichuan Province 2021ZHYZ0023National Natural Science Foundation of China 82070660
6 · The paper itself

Abstract

Frasier syndrome (FS) is a rare inherited disorder characterized by gonadal dysgenesis and progressive nephropathy, resulting from mutations in the intron 9 splice donor site of the Wilms tumor 1 (WT1) gene. It is associated with male gonadal dysgenesis (female external genitalia with a 46 XY karyotype), and a high risk of gonadoblastoma during adolescence. Patients with FS present early in childhood with proteinuria that progressively worsens with a high likelihood of end-stage renal disease (ESRD). Herein, we report a 15-year-old female (karyotype 46, XY) patient characterized by delayed puberty and steroid-resistant nephrotic syndrome, in whom whole genome sequencing showed a mutation in intron 9 of the WT1 gene, c.1447 + 4 C>T. This is the first case of FS with delayed puberty as the first complaint with no previous renal symptoms. We consider delayed puberty as an important manifestation of FS and summarize the diagnostic process of delayed puberty in the female phenotype. For clinicians, delayed puberty is a common disorder in pediatrics but requires vigilance for some rare causes. Etiological screening and chromosome karyotype analysis are important for the early diagnosis of FS in patients with delayed puberty.

Indexed as

case reportdelayed pubertyfocal segmental glomerulosclerosisFrasier syndromenephrotic syndromepediatricsWilms tumor 1

Identifiers

PMID36980135
PMCPMC10046944
OpenAlexW4327741205

What OpenQuestion holds

Textfull text, public
LicenceCC BY
measurements read10
identifiers read2
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.