Evidence map›Paper›PMID 33369061›Full record

ArticleAmerican journal of medical genetics. Part A2021

Further delineation of a recognizable type of syndromic short stature caused by biallelic SEMA3A loss-of-function variants.

Alexander F Gileta, Maria L Helgeson, Jacqueline M M Leonard, Louise C Pyle, Hari P Subramanian, Kelly Arndt, Colin P Hawkes, Daniela Del Gaudio

Open access · greenAbstract readCase Reports
In one paragraph

Article in American journal of medical genetics. Part A, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.3field-weighted citation impact, top 47% 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

3 citing papers in PubMed, 3 citations in OpenAlex.

  1. Review
  2. Article
  3. 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 2 institutions in 1 country.

Alexander F GiletaDepartment of Human Genetics, The University of Chicago, Chicago, Illinois, USA.ORCID 0000-0002-7793-8043
Maria L HelgesonDepartment of Human Genetics, The University of Chicago, Chicago, Illinois, USA.
Jacqueline M M LeonardDivision of Human Genetics and Roberts Individualized Medical Genetics Center, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
Louise C PyleDivision of Human Genetics and Roberts Individualized Medical Genetics Center, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.ORCID 0000-0002-8933-9320
Hari P SubramanianDepartment of Human Genetics, The University of Chicago, Chicago, Illinois, USA.
Kelly ArndtDepartment of Human Genetics, The University of Chicago, Chicago, Illinois, USA.
Colin P HawkesPerelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania, USA.
Daniela Del GaudioDepartment of Human Genetics, The University of Chicago, Chicago, Illinois, USA.
University of Chicago · USChildren's Hospital of Philadelphia · US

Funding

Institutional Clinical and Translational Science AwardKL2TR001879 · NCATS · UNIVERSITY OF PENNSYLVANIA · PI MEAGHER, EMMA ANNE · 2016 to 2025
$13.7M
MEDICAL GENETICS RESEARCH TRAINING GRANTT32GM008638 · NIGMS · UNIVERSITY OF PENNSYLVANIA · PI Rebecca Ganetzky · 1997 to 2026
$8.7M
NCATS NIH HHS KL2 TR001879NIGMS NIH HHS T32 GM008638
6 · The paper itself

Abstract

The semaphorin protein family is a diverse set of extracellular signaling proteins that perform fundamental roles in the development and operation of numerous biological systems, notably the nervous, musculoskeletal, cardiovascular, endocrine, and reproductive systems. Recently, recessive loss-of-function (LoF) variants in SEMA3A (semaphorin 3A) have been shown to result in a recognizable syndrome characterized by short stature, skeletal abnormalities, congenital heart defects, and variable additional anomalies. Here, we describe the clinical and molecular characterization of a female patient presenting with skeletal dysplasia, hypogonadotropic hypogonadism (HH), and anosmia who harbors a nonsense variant c.1633C>T (p.Arg555*) and a deletion of exons 15, 16, and 17 in SEMA3A in the compound heterozygous state. These variants were identified through next-generation sequencing analysis of a panel of 26 genes known to be associated with HH/Kallmann syndrome. Our findings further substantiate the notion that biallelic LoF SEMA3A variants cause a syndromic form of short stature and expand the phenotypic spectrum associated with this condition to include features of Kallmann syndrome.

Indexed as

Codon, NonsenseLoss of Function MutationAbnormalities, MultipleAllelesAnosmiaClubfootDwarfismFemaleHeart Defects, CongenitalHeterozygoteHigh-Throughput Nucleotide SequencingHumansHypogonadismInfant, NewbornKallmann SyndromeMuscle HypotoniaCodon, NonsenseSEMA3A protein, humanSemaphorin-3Aanosmiahypogonadotropic hypogonadismKallmann syndromeSEMA3Ashort stature

Identifiers

PMID33369061
PMCPMC10258807
OpenAlexW3118135535

What OpenQuestion holds

Textmetadata
LicenceTDM
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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.