Evidence map›Paper›PMID 36541232›Full record

ArticleAmerican journal of medical genetics. Part A2023

Patterns of co-occurring birth defects in children with anotia and microtia.

Jeremy M Schraw, Renata H Benjamin, Charles J Shumate, Mark A Canfield, Daryl A Scott, Scott D McLean, Hope Northrup, Angela E Scheuerle, Christian P Schaaf, Joseph W Ray and 3 more

Open access · greenAbstract read
In one paragraph

Article in American journal of medical genetics. Part A, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.5field-weighted citation impact, top 33% 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

2 citing papers in PubMed, 3 citations in OpenAlex.

  1. Article
  2. 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

13 authors at 7 institutions in 2 countries.

Jeremy M SchrawDepartment of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.
Renata H BenjaminDepartment of Epidemiology, Human Genetics & Environmental Sciences, UTHealth School of Public Health, Houston, Texas, USA.ORCID 0000-0001-6406-4247
Charles J ShumateBirth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, Texas, USA.
Mark A CanfieldBirth Defects Epidemiology and Surveillance Branch, Texas Department of State Health Services, Austin, Texas, USA.ORCID 0000-0001-8827-1881
Daryl A ScottDepartment of Molecular and Human Genetics, Baylor College of Medicine, Houston, Texas, USA.ORCID 0000-0003-1460-5169
Scott D McLeanDepartment of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.
Hope NorthrupDepartment of Pediatrics, Division of Medical Genetics, McGovern Medical School at the University of Texas Health Science Center at Houston (UTHealth), Houston, Texas, USA.
Angela E ScheuerleDepartment of Pediatrics, University of Texas Southwestern Medical Center, Dallas, Texas, USA.ORCID 0000-0002-9025-3276
Christian P SchaafInstitute of Human Genetics, Heidelberg University, Heidelberg, Germany.ORCID 0000-0002-2148-7490
Joseph W RayDepartment of Pediatrics, Division of Medical Genetics and Metabolism, University of Texas Medical Branch, Galveston, Texas, USA.
Han ChenDepartment of Epidemiology, Human Genetics & Environmental Sciences, UTHealth School of Public Health, Houston, Texas, USA.
A J AgopianDepartment of Epidemiology, Human Genetics & Environmental Sciences, UTHealth School of Public Health, Houston, Texas, USA.ORCID 0000-0002-5874-4155
Philip J LupoDepartment of Pediatrics, Baylor College of Medicine, Houston, Texas, USA.
Baylor College of Medicine · USUniversity of Texas Health Science Center at Dallas · USTexas Department of State Health Services · USHeidelberg University · DEMemorial Hermann · USThe University of Texas Medical Branch at Galveston · USThe University of Texas Southwestern Medical Center · US

Funding

A multidisciplinary approach for identifying and characterizing novel congenital malformation syndromesR01HD093660 · NICHD · UNIVERSITY OF TEXAS HLTH SCI CTR HOUSTON · PI AGOPIAN, A.J., LUPO, PHILIP · 2018 to 2022
$2.0M
NICHD NIH HHS R01 HD093660
6 · The paper itself

Abstract

Many infants with anotia or microtia (A/M) have co-occurring birth defects, although few receive syndromic diagnoses in the perinatal period. Evaluation of co-occurring birth defects in children with A/M could identify patterns indicative of undiagnosed/unrecognized syndromes. We obtained information on co-occurring birth defects among infants with A/M for delivery years 1999-2014 from the Texas Birth Defects Registry. We calculated observed-to-expected ratios (OER) to identify birth defect combinations that occurred more often than expected by chance. We excluded children diagnosed with genetic or chromosomal syndromes from analyses. Birth defects and syndromes/associations diagnosed ≤1 year of age were considered. We identified 1310 infants with non-syndromic A/M, of whom 38% (N = 492) were diagnosed with co-occurring major defects. Top combinations included: hydrocephalus, ventricular septal defect, and spinal anomalies (OER 58.4); microphthalmia and anomalies of the aorta (OER 55.4); and cleft lip with or without cleft palate and rib or sternum anomalies (OER 32.8). Some combinations observed in our study may represent undiagnosed/atypical presentations of known A/M associations or syndromes, or novel syndromes yet to be described in the literature. Careful evaluation of infants with multiple birth defects including A/M is warranted to identify individuals with potential genetic or chromosomal syndromes.

Indexed as

Abnormalities, MultipleCleft LipCleft PalateCongenital AbnormalitiesCongenital MicrotiaFemaleHumansInfantPregnancyTexasanotiaco-occurring defectsepidemiologymicrotia

Identifiers

PMID36541232
PMCPMC9928897
OpenAlexW4312066245

What OpenQuestion holds

Textmetadata
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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.