Evidence map›Paper›PMID 35986191›Full record

ArticleJournal of community genetics2022

Experiences of individuals receiving a sex chromosome multisomy diagnosis.

Jordan P Richardson, Nivedita Ahlawat, Kirsten A Riggan, Sharron Close, Megan A Allyse

Open access · greenAbstract read
In one paragraph

Article in Journal of community genetics, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
1.8field-weighted citation impact, top 15% 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

5 citing papers in PubMed, 6 citations in OpenAlex.

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  4. Review
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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

5 authors at 5 institutions in 1 country.

Jordan P RichardsonBiomedical Ethics Research Program, Mayo Clinic, Rochester, MN, USA.ORCID http://orcid.org/0000-0002-5795-5241
Nivedita AhlawatDepartment of Genetics, Stanford University School of Medicine, Stanford, CA, USA.
Kirsten A RigganBiomedical Ethics Research Program, Mayo Clinic, Rochester, MN, USA.ORCID http://orcid.org/0000-0002-0127-9555
Sharron CloseNell Hodgson Woodruff School of Nursing, Emory University, Atlanta, GA, USA.ORCID http://orcid.org/0000-0002-2117-2165
Megan A AllyseBiomedical Ethics Research Program, Mayo Clinic, Rochester, MN, USA. Allyse.megan@mayo.edu.ORCID http://orcid.org/0000-0001-6136-9256
Emory University · USJacksonville College · USMayo Clinic · USMayo Clinic in Florida · USStanford University · US

Funding

Mayo Clinic Center for Clinical and Translational Science (CCaTS UL1 Supplement - Dr. Timothy Curry)UL1TR002377 · NCATS · MAYO CLINIC ROCHESTER · PI VESNA D GAROVIC · 2017 to 2026
$78.4M
Factors Influencing Access and Utilization of Genetic Prenatal Care Services Among Women from Underserved PopulationsK01HG009542 · NHGRI · MAYO CLINIC ROCHESTER · PI ALLYSE, MEGAN A. · 2018 to 2022
$809k
NCATS NIH HHS UL1 TR002377NHGRI NIH HHS K01 HG009542
6 · The paper itself

Abstract

Sex chromosome multisomies (SCMs) are genomic conditions with variable phenotypes that range from undetectable to requiring extensive clinical intervention. Currently, many individuals with SCMs are diagnosed in adolescence or adulthood based on physical symptoms related to pubertal development and infertility. Given the expansion of genetic testing in routine clinical practice, the diagnosing clinician is increasingly a primary care or family medicine provider. This study aims to help providers better understand the patient experience of receiving a diagnosis. We conducted a survey of individuals (n = 55) with SCMs using closed and open-ended questions. Open-response questions were qualitatively analyzed and are reported here with the descriptive results of the closed-ended questions. Most participants were diagnosed with 47,XXY (n = 51; 85.0%), identified as White race/ethnicity (n = 46; 88.5%), and were college graduates or higher (n = 29; 54.7%). Many participants reported dissatisfaction with the delivery of the diagnosis, expressing that it was rushed and their provider lacked detailed information about the condition. Participants were frustrated by the general lack of availability of high-quality informational resources from both medical and other sources at the time of diagnosis. Some participants also described the social and psychological impact of the diagnosis and how it was delivered. To the best of our knowledge, this is the largest survey of individuals diagnosed with SCMs, which is notable considering their prevalence and low diagnostic rate. Our findings provide patient-informed insight on how to improve the delivery of SCM diagnoses, especially delivery in a primary care setting, including the provision of up-to-date information and proactive referral to specialty care and counseling services.

Indexed as

Clinical encounterGenetic diagnosisKlinefelter syndromePsychosocial supportSex chromosome aneuploidy

Identifiers

PMID35986191
PMCPMC9681968
OpenAlexW4294250176

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.