Evidence map›Paper›PMID 39502852›Full record

ArticleJournal of pediatric genetics2024

Novel Case of Prader-Willi Syndrome and Ebstein's Anomaly: Implications for Complex Care Management.

Donald Mattia, Christopher Lindblade, Oliver Oatman, Supraja Prakash, Theresa Grebe

Abstract read
In one paragraph

Article in Journal of pediatric genetics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
–field-weighted citation impact
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

0 citing papers in PubMed.

No citing paper in PubMed yet.

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.

Donald MattiaDepartment of General Pediatrics, Phoenix Children's Hospital, Phoenix, Arizona, United States.ORCID 0000-0002-0440-6249
Christopher LindbladeDepartment of Cardiology, Phoenix Children's Hospital, Phoenix, Arizona, United States.
Oliver OatmanDepartment of Endocrinology, Phoenix Children's Hospital, Phoenix, Arizona, United States.
Supraja PrakashDepartment of Genetics and Metabolism, Phoenix Children's Hospital, Phoenix, Arizona, United States.
Theresa GrebeDepartment of Genetics and Metabolism, Phoenix Children's Hospital, Phoenix, Arizona, United States.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

We present a patient with a complex phenotype including diagnoses of Ebstein's anomaly and Prader-Willi syndrome (PWS) as well as additional congenital anomalies and genetic variants with potential clinical effects. This is the first reported case of both diagnoses present in the same patient. The diagnosis of Ebstein's anomaly was made on prenatal ultrasound. She presented with neonatal hypotonia, feeding problems, and dysmorphic features, followed by later onset weight gain, leading to a diagnosis of PWS. Further evaluations revealed Blaschkoid hyperpigmentation, laryngeal cleft, and pigmentary retinopathy. Whole exome sequencing determined a likely pathogenic variant in alkaline phosphatase gene and several mitochondrial DNA variants. We discuss the known genetic mechanisms of PWS and compare them to the heterogenous genetic associations of Ebstein's anomaly. The standard of care treatment for PWS is growth hormone therapy, which is associated with right-sided heart failure risks. This case illustrates the need to complete the diagnostic work up in all patients, as well as the necessity of a multidisciplinary approach for optimal outcomes.

Indexed as

Blaschkoid hyperpigmentationEbstein's anomalyGrowth hormonePrader–Willi

Identifiers

PMID39502852
PMCPMC11534433

What OpenQuestion holds

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Registered trials

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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.