Evidence map›Paper›PMID 23269230›Full record

ArticleJournal of perinatology : official journal of the California Perinatal Association2013

Erroneous prenatal diagnosis of congenital adrenal hyperplasia owing to a duplication of the CYP21A2 gene.

O Lekarev, K Tafuri, A H Lane, G Zhu, J M Nakamoto, A M Buller-Burckle, T A Wilson, M I New

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In one paragraph

Article in Journal of perinatology : official journal of the California Perinatal Association, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers.

0numbers the graph read from it
0cells of the map it votes in
15citing papers in PubMed
0.8field-weighted citation impact, top 29% 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

15 citing papers in PubMed, 21 citations in OpenAlex.

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  13. The next 150 years of congenital adrenal hyperplasia.The Journal of steroid biochemistry and molecular biology · 2015
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  14. Review
  15. Genotype-phenotype correlation in 1,507 families with congenital adrenal hyperplasia owing to 21-hydroxylase deficiency.Proceedings of the National Academy of Sciences of the United States of America · 2013
    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

8 authors at 3 institutions in 1 country.

O LekarevDepartment of Pediatric Endocrinology, Adrenal Steroid Disorders Program, Mount Sinai School of Medicine, New York, NY 10029, USA.
K Tafuri
A H Lane
G Zhu
J M Nakamoto
A M Buller-Burckle
T A Wilson
M I New
Stony Brook University Hospital · USIcahn School of Medicine at Mount Sinai · USQuest Diagnostics (United States) · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Congenital adrenal hyperplasia (CAH) is an autosomal recessive disorder where steroidogenesis in the adrenal cortex is impaired. The most common form is caused by 21-hydroxylase deficiency (21OHD). Classical 21OHD is characterized by glucocorticoid and mineralocorticoid deficiency and by overproduction of adrenal androgens. The diagnosis rests on biochemical and genetic analyses. In families with history of CAH, prenatal genetic diagnosis is offered. We herein present a case of an infant whose parents were identified to carry mutations on the CYP21A2 gene. The fetal DNA analysis demonstrated that the fetus carried a paternal exon 8 (Q318X) mutation and a maternal exon 8 (R356X) mutation. The fetus was presumed to be affected with CAH, yet his clinical presentation at birth was not consistent with the diagnosis. Repeated genetic analysis identified a paternal CYP21A2 gene duplication with Q318X mutation on one copy of CYP21A2. We conclude that a duplication of the CYP21A2 gene should be suspected when clinical and hormonal findings do not support the genetic diagnosis. Furthermore, because individuals with Q318X mutation frequently have a duplication of the CYP21A2 gene, when Q318X is detected, it is important to distinguish the severe point mutation in single gene copy alleles from the non-deficient variant in gene-duplicated alleles.

Indexed as

Diagnostic ErrorsPrenatal Diagnosis17-alpha-HydroxyprogesteroneAdrenal Hyperplasia, CongenitalAdultChorionic Villi SamplingDNA Mutational AnalysisExonsFemaleFollow-Up StudiesGene DuplicationGenetic Carrier ScreeningGenetic TestingHapMap ProjectHumansInfant17-alpha-HydroxyprogesteroneCYP21A2 protein, humanReninSteroid 21-Hydroxylase

Identifiers

PMID23269230
OpenAlexW2055990049

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.