Evidence map›Paper›PMID 34487203›Full record

ArticleCalcified tissue international2022

Two De Novo Mosaic Variants Within the Same Site of PHEX Gene in a Girl with X-Linked Hypophosphatemic Rickets.

Yunting Lin, Wen Zhang, Xinjiang Huang, Ling Su, Yanna Cai, Cuili Liang, Min Rao, Li Liu, Chunhua Zeng

Abstract readCase Reports
PubMed Publisher
In one paragraph

Article in Calcified tissue international, 2022. 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
0.3field-weighted citation impact, top 43% 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. Article
  2. UnusualJBMR plus · 2025
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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.

Yunting LinDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.ORCID 0000-0001-8974-357X
Wen ZhangDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.
Xinjiang HuangDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.
Ling SuDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.
Yanna CaiDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.
Cuili LiangDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.
Min RaoDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China.
Li LiuDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China. liliuchina@qq.com.ORCID 0000-0002-4330-5694
Chunhua ZengDepartment of Genetics and Endocrinology, Guangzhou Women and Children's Medical Center, Guangzhou Medical University, 9 Jinsui Rd., Guangzhou, 510623, China. chunhuazeng@hotmail.com.ORCID 0000-0001-5954-1803
Guangzhou Medical University · CNGuangzhou Women and Children Medical Center · CN

Funding

guangzhou science and technology program 201804010283national natural science foundation of china 81873661special fund of guangzhou science technology and innovation commission 202102020133
6 · The paper itself

Abstract

X-linked hypophosphatemic rickets (XLH) is the most common form of hypophosphatemic rickets, which is caused by the deficiencies of PHEX gene with an X-linked dominant inheritance pattern. As at least several thousands of XLH patients have been diagnosed, only several males and fewer females with mosaicism of PHEX gene were found. Here we describe an XLH girl with two de novo mosaic variants within the same site of PHEX gene. To rapidly screen all of the causative genes of hypophosphatemic rickets and rule out other diseases, DNA samples were initially analyzed using whole exome sequencing (WES). Interestingly, two different pathogenic mosaic variants, a known c.1809G > A(p.W603*) variant and a novel c.1809G > T(p.W603C) variant within the same site of PHEX gene, were identified in the proband by WES. Subsequent Sanger sequencing confirmed the presence and de novo pattern of these two mosaic variants in the proband, which were absent in her healthy parents. This is the first case to report two different mosaic variants of PHEX gene in an XLH individual. This XLH girl has a de novo mosaic genotype of c.1809 = /G > T/G > A in PHEX gene. Our report adds an unusual mocaicism case for XLH and expands the mutational event and spectrum of PHEX gene. Our report also alerts clinicians and geneticists to be cautious about mocaicism and detection methods.

Indexed as

Familial Hypophosphatemic RicketsGenetic Diseases, X-LinkedPHEX Phosphate Regulating Neutral EndopeptidaseFemaleGenotypeHumansMaleMutationPHEX Phosphate Regulating Neutral EndopeptidasePHEX protein, humanMosaicismPHEX geneSanger sequencingWhole exome sequencingX-linked hypophosphatemic rickets

Identifiers

PMID34487203
OpenAlexW3197455009

What OpenQuestion holds

Textmetadata
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.