Evidence map›Paper›PMID 36308388›Full record

ArticleAmerican journal of medical genetics. Part A2023

Obstetrical and neonatal outcomes of cardio-facio-cutaneous syndrome: Prenatal consequences of Ras/MAPK dysregulation.

Angie C Jelin, Amanda Mahle, Susan H Tran, Teresa N Sparks, Katherine A Rauen

Open access · greenAbstract read
In one paragraph

Article in American journal of medical genetics. Part A, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.4field-weighted citation impact, top 41% 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

1 citing paper in PubMed, 5 citations in OpenAlex.

  1. 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

5 authors at 4 institutions in 1 country.

Angie C JelinDepartment of Gynecology and Obstetrics, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States.ORCID 0000-0002-1792-4029
Amanda MahleDepartment of Gynecology and Obstetrics, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States.
Susan H TranDivision of Maternal-Fetal Medicine, Legacy Medical Group, Portland, Oregon, United States.
Teresa N SparksDepartment of Obstetrics, Gynecology and Reproductive Sciences, University of California San Francisco, San Francisco, California, United States.
Katherine A RauenDivision of Genomic Medicine, Department of Pediatrics, MIND Institute, University of California Davis, Davis, California, United States.ORCID 0000-0003-1719-7228
Johns Hopkins University · USLegacy Emanuel Medical Center · USUniversity of California, Davis · USUniversity of California, San Francisco · US

Funding

WOMEN'S REPRODUCTIVE HEALTH RESEARCH CENTERK12HD001262 · NICHD · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Andrea Vashti Jackson · 1998 to 2026
$9.3M
Uncovering the etiologies of non-immune hydrops fetalis through comprehensive genomic analyses and phenotypingR01HD107190 · NICHD · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI Teresa N Sparks · 2022 to 2026
$3.6M
Single gene pathogenic variants associated with BEEC (Bladder Exstrophy, Epispadias, Complex)K23DK119949 · NIDDK · JOHNS HOPKINS UNIVERSITY · PI JELIN, ANGIE CHILD · 2019 to 2023
$1.1M
NICHD NIH HHS K12 HD001262NICHD NIH HHS R01 HD107190NIDDK NIH HHS K23 DK119949
6 · The paper itself

Abstract

We systematically delineated the prenatal phenotype, and obstetrical and neonatal outcomes of the RASopathy cardio-facio-cutaneous (CFC) syndrome. A comprehensive, retrospective medical history survey was distributed to parents of children with confirmed CFC in collaboration with CFC International, Inc. Data were collected on CFC gene variant, maternal characteristics, pregnancy course, delivery, and neonatal outcomes with the support of medical records. We identified 43 individuals with pathogenic variants in BRAF (81%), MEK1 (14%), or MEK2 (5%) genes. The median age was 8.5 years. Hyperemesis gravidarum, gestational diabetes, gestational hypertension, and preeclampsia occurred in 5/43 (12%), 4/43 (9%), 3/43 (7%), and 3/43 (7%) of pregnancies, respectively. Second and third trimester ultrasound abnormalities included polyhydramnios, macrocephaly, macrosomia, and renal and cardiac abnormalities. Delivery occurred via spontaneous vaginal, operative vaginal, or cesarean delivery in 15/42 (36%), 7/42 (16%), and 20/42 (48%), respectively. Median gestational age at delivery was 37 weeks and median birth weight was 3501 grams. Germline pathogenic vaiants had mutiple congenital consequences including polyhydramnios, renal and cardiac abnormalities, macrosomia, and macrocephaly on second and third trimester ultrasound. Elevated rates of operative delivery and neonatal complications were also noted. Understanding and defining a prenatal phenotype may improve prenatal prognostic counseling and outcomes.

Indexed as

Ectodermal DysplasiaHeart Defects, CongenitalMegalencephalyPolyhydramniosFaciesFailure to ThriveFemaleFetal MacrosomiaHumansPregnancyProto-Oncogene Proteins B-rafRetrospective StudiesProto-Oncogene Proteins B-rafcardio-facio-cutaneous syndromeprenatalRas/MAPKRASopathysignal transduction pathway

Identifiers

PMID36308388
PMCPMC9839479
OpenAlexW4307532263

What OpenQuestion holds

Textmetadata
LicenceTDM
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.