Evidence map›Paper›PMID 36598290›Full record

ReviewEndocrine connections2023

New developments and future trajectories in supernumerary sex chromosome abnormalities: a summary of the 2022 3rd International Workshop on Klinefelter Syndrome, Trisomy X, and XYY.

Claus H Gravholt, Alberto Ferlin, Joerg Gromoll, Anders Juul, Armin Raznahan, Sophie van Rijn, Alan D Rogol, Anne Skakkebæk, Nicole Tartaglia, Hanna Swaab

Open access · goldAbstract readReview
In one paragraph

Review in Endocrine connections, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.

0numbers the graph read from it
0cells of the map it votes in
6citing papers in PubMed
3.8field-weighted citation impact, top 7% 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

6 citing papers in PubMed, 12 citations in OpenAlex.

  1. Article
  2. Differences of sex development.Nature reviews. Disease primers · 2025
    Review
  3. Update on Physical, Psychological, and Quality of Life Management in Klinefelter Syndrome.The Journal of clinical endocrinology and metabolism · 2025
    Review
  4. Review
  5. Article
  6. 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

10 authors at 7 institutions in 4 countries.

Claus H GravholtDepartment of Endocrinology, Aarhus University Hospital, Aarhus, Denmark.ORCID 0000-0001-5924-1720
Alberto FerlinDepartment of Medicine, Unit of Andrology and Reproductive Medicine, University of Padova, Padova, Italy.
Joerg GromollCentre of Reproductive Medicine and Andrology, Münster, Germany.
Anders JuulDepartment of Growth and Reproduction Copenhagen University Hospital - Rigshospitalet, Copenhagen, Denmark.ORCID 0000-0002-0534-4350
Armin RaznahanSection on Developmental Neurogenomics, National Institute of Mental Health Intramural Research Program, National Institutes of Health, Bethesda, Maryland, USA.
Sophie van RijnClinical Neurodevelopmental Sciences, Leiden University, Leiden, The Netherlands and TRIXY Center of Expertise, Leiden University Treatment and Expertise Centre (LUBEC), Leiden, The Netherlands.ORCID 0000-0002-9179-7515
Alan D RogolDepartment of Pediatrics, University of Virginia, Charlottesville, Virginia, USA.ORCID 0000-0002-7526-3142
Anne SkakkebækDepartment of Molecular Medicine, Aarhus University Hospital, Aarhus, Denmark.ORCID 0000-0001-9178-4901
Nicole TartagliaDepartment of Pediatrics, Developmental Pediatrics, University of Colorado School of Medicine, Aurora, Colorado, USA.
Hanna SwaabClinical Neurodevelopmental Sciences, Leiden University, Leiden, The Netherlands and TRIXY Center of Expertise, Leiden University Treatment and Expertise Centre (LUBEC), Leiden, The Netherlands.
Aarhus University · DKLeiden University · NLNational Institutes of Health · USUniversity of Colorado Denver · USUniversity of Copenhagen · DKUniversity of Padua · ITUniversity of Virginia · US

Funding

Section on Developmental NeurogenomicsZIAMH002949 · NIMH · NATIONAL INSTITUTE OF MENTAL HEALTH · PI RAZNAHAN, ARMIN · 2016 to 2025
$30.0M
The eXtraordinarY Babies Study: Natural History of Health and Neurodevelopment in Infants with Sex Chromosome TrisomyR01HD091251 · NICHD · UNIVERSITY OF COLORADO DENVER · PI Nicole Renee Tartaglia · 2017 to 2026
$5.6M
NCBDD CDC HHS U01 DD001298NICHD NIH HHS R01 HD091251
6 · The paper itself

Abstract

The 3rd International Workshop on Klinefelter Syndrome, Trisomy X, and 47,XYY syndrome was held in Leiden, the Netherlands, on September 12-14, 2022. Here, we review new data presented at the workshop and discuss scientific and clinical trajectories. We focus on shortcomings in knowledge and therefore point out future areas for research. We focus on the genetics and genomics of supernumerary sex chromosome syndromes with new data being presented. Most knowledge centre specifically on Klinefelter syndrome, where aspects on testosterone deficiency and the relation to bone, muscle and fat were discussed, as was infertility and the treatment thereof. Both trisomy X and 47,XYY syndrome are frequently affected by infertility. Transitioning of males with Klinefelter syndrome was addressed, as this seemingly simple process in practise is often difficult. It is now realized that neurocognitive changes are pervasive in all supernumerary sex chromosome syndromes, which were extensively discussed. New intervention projects were also described, and exciting new data concerning these were presented. Advocacy organizations were present, describing the enormous burden carried by parents when having to explain their child's specific syndrome to most professionals whenever in contact with health care and education systems. It was also pointed out that most countries do not have health care systems that diagnose patients with supernumerary sex chromosome syndromes, thus pinpointing a clear deficiency in the current genetic testing and care models. At the end of the workshop, a roadmap towards the development of new international clinical care guidelines for Klinefelter syndrome was decided.

Indexed as

47,XYY syndromeanti-Mullerian hormoneKlinefelter syndrometestosteronetrisomy X syndrome

Identifiers

PMID36598290
PMCPMC9986408
OpenAlexW4313488932

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
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.