Evidence map›Paper›PMID 30079495›Full record

ArticleAmerican journal of medical genetics. Part A2018

Dual diagnoses in 152 patients with Turner syndrome: Knowledge of the second condition may lead to modification of treatment and/or surveillance.

Kelly L Jones, Erin A McNamara, Mauro Longoni, Danny E Miller, Mersedeh Rohanizadegan, Laura A Newman, Frances Hayes, Lynne L Levitsky, Betty L Herrington, Angela E Lin

Open access · greenAbstract readCase Reports
In one paragraph

Article in American journal of medical genetics. Part A, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed
1.3field-weighted citation impact, top 18% 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

12 citing papers in PubMed, 21 citations in OpenAlex.

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  12. Select pediatric vitreoretinal disease in the setting of Turner's syndrome.American journal of ophthalmology case reports · 2020
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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

10 authors at 5 institutions in 1 country.

Kelly L JonesDivision of Medical Genetics and Metabolism, Children's Hospital of The King's Daughters, Norfolk, Virginia.ORCID 0000-0002-5097-9865
Erin A McNamaraGenetics Unit, MassGeneral Hospital for Children, Boston, Massachusetts.
Mauro LongoniPediatric Surgical Research Laboratories, Massachusetts General Hospital, Boston, Massachusetts.
Danny E MillerDepartment of Pediatrics, Seattle Children's Hospital, University of Washington, Seattle, Washington.
Mersedeh RohanizadeganDivision of Genetics and Genomics, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts.
Laura A NewmanDivision of Pediatric Hematology/Oncology, Department of Pediatrics, University of Mississippi, Jackson, Mississippi.
Frances HayesDepartment of Medicine, Reproductive Endocrinology, Massachusetts General Hospital, Boston, Massachusetts.
Lynne L LevitskyPediatric Endocrinology, MassGeneral Hospital for Children, Boston, Massachusetts.
Betty L HerringtonDivision of Pediatric Hematology/Oncology, Department of Pediatrics, University of Mississippi, Jackson, Mississippi.
Angela E LinGenetics Unit, MassGeneral Hospital for Children, Boston, Massachusetts.
Boston Children's Hospital · USMassachusetts General Hospital · USUniversity of Mississippi Medical Center · USEastern Virginia Medical School · USSeattle Children's Hospital · US

Funding

TRAINING GRANT IN GENETICST32GM007748 · NIGMS · HARVARD UNIVERSITY (MEDICAL SCHOOL) · PI Anne O'Donnell-Luria, Louise Wilkins-Haug · 1985 to 2026
$12.0M
NIGMS NIH HHS T32 GM007748
6 · The paper itself

Abstract

Turner syndrome is a sex chromosome abnormality in which a female has a single X chromosome or structurally deficient second sex chromosome. The phenotypic spectrum is broad, and atypical features prompt discussion of whether the known features of Turner syndrome should be further expanded. With the advent of clinical whole exome sequencing, there has been increased realization that some patients with genetic disorders carry a second genetic disorder, leading us to hypothesize that a "dual diagnosis" may be more common than suspected for Turner syndrome. We report five new patients with Turner syndrome and a co-occurring genetic disorder including one patient with Li-Fraumeni syndrome, Li-Fraumeni and Noonan syndrome, mosaic trisomy 8, pathogenic variant in RERE, and blepharophimosis-ptosis-epicanthanus inversus syndrome. We also undertook an extensive literature review of 147 reports of patients with Turner syndrome and a second genetic condition. A total of 47 patients (31%) had trisomy 21, followed by 36 patients (24%) had one of 11 X-linked disorders. Notably, 80% of the 147 reported patients with a dual diagnosis had mosaicism for Turner syndrome, approximately twice the frequency in the general Turner syndrome population. This article demonstrates the potential for co-occurring syndromes in patients with Turner syndrome, prompting us to recommend a search for an additional genetic disorder in Turner patients with unusual features. Knowledge of the second condition may lead to modification of treatment and/or surveillance. We anticipate that increased awareness and improved diagnostic technologies will lead to the identification of more cases of Turner syndrome with a co-occurring genetic syndrome.

Indexed as

Population SurveillanceAdultChildChild, PreschoolFemaleHumansTurner Syndromeblepharophimosis-ptosis-epicanthus inversusdual diagnosisLi-Fraumeni syndromeNoonan syndromeRERE genesex chromosome abnormality syndrometrisomy 8Turner syndromewhole exome sequencing

Identifiers

PMID30079495
PMCPMC6289717
OpenAlexW2887015344

What OpenQuestion holds

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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.