Evidence map›Paper›PMID 37186369›Full record

ArticleBirth defects research2023

A multi-program analysis of cleft lip with cleft palate prevalence and mortality using data from 22 International Clearinghouse for Birth Defects Surveillance and Research programs, 1974-2014.

Niall Mc Goldrick, Gavin Revie, Boris Groisman, Paula Hurtado-Villa, Antonin Sipek, Babak Khoshnood, Anke Rissmann, Saeed Dastgiri, Danielle Landau, Giovanna Tagliabue and 15 more

Open access · hybridAbstract read
In one paragraph

Article in Birth defects research, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 13 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
13citing papers in PubMed, 1 pooled it
6.9field-weighted citation impact, top 3% 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

13 citing papers in PubMed, 1 synthesis or guideline pooled it, 22 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Prerepair Mortality in Cleft Lip and/or Palate: A Retrospective Analysis of Early Childhood Deaths.The Cleft palate-craniofacial journal : official publication of the American Cleft Palate-Craniofacial Association · 2026
    Article
  4. Feasibility of Imaging the Uvula at the Midtrimester Anomaly Ultrasound: A Retrospective Study.Journal of ultrasound in medicine : official journal of the American Institute of Ultrasound in Medicine · 2026
    Article
  5. Article
  6. Article
  7. Article
  8. Mortality in Cleft Lip and Palate Patients: A Systematic Review and Meta-analysis.Plastic and reconstructive surgery. Global open · 2025
    Article
  9. Article
  10. Article
  11. Prevalence of Congenital Anomalies in Iran.Archives of Iranian medicine · 2024
    Article
  12. Article
  13. 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

25 authors at 20 institutions in 15 countries.

Niall Mc GoldrickSchool of Dentistry, University of Dundee, Dundee, Scotland, UK.ORCID 0000-0001-7353-1396
Gavin RevieSchool of Dentistry, University of Dundee, Dundee, Scotland, UK.
Boris GroismanNational Network of Congenital Anomalies of Argentina (RENAC), National Institute of Epidemiology (INE), ANLIS, National Ministry of Health, National Center of Medical Genetics, Ciudad de Buenos Aires, Argentina.ORCID 0000-0001-6263-2562
Paula Hurtado-VillaFacultad de Ciencias de la Salud, Pontificia Universidad Javeriana, Cali, Colombia.
Antonin SipekDepartment of Medical Genetics, Thomayer University Hospital, Prague, Czech Republic.
Babak KhoshnoodUniversité Paris Cité, Inserm, INRAE, Centre for Research in Epidemiology and StatisticS (CRESS), Obstetrical Perinatal and Pediatric Epidemiology Research Team, EPOPé, Paris, France.
Anke RissmannMalformation Monitoring Centre Saxony-Anhalt, Medical Faculty Otto-von-Guericke-University Magdeburg, Magdeburg, Germany.ORCID 0000-0002-9437-2790
Saeed DastgiriTabriz Health Services Management Research Center, Tabriz University of Medical Sciences, Tabriz, Iran.
Danielle LandauIsrael Birth Defects Surveillance Program, NICU Soroka University Medical Center, Beer Sheva, Israel.
Giovanna TagliabueCancer Registry Unit, Fondazione IRCCS Istituto Nazionale dei Tumori, Lombardy birth defects registry, Lombardy, Italy.
Anna PieriniInstitute of Clinical Physiology, National Research Council and Fondazione Toscana Gabriele Monasterio, Tuscany Registry of Congenital Defects, Pisa, Italy.
Miriam GattMalta Congenital Anomalies Registry, Directorate for Health Information and Research, Tal-Pietà, Malta.
Osvaldo M MutchinickRegistry and Epidemiological Surveillance of Congenital Malformations (RYVEMCE), National Institute of Medical Sciences and Nutrition, Mexico City, Mexico.
Laura MartínezGenetics Department University Hospital, Universidad Autonoma de Nuevo Leon, Nuevo Leon, Mexico.
Hermein E K de WalleDepartment of Genetics, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands.
Elena SzabovaFaculty of Public Health, Slovak Medical University, Bratislava, Slovakia.
Jorge Lopez CameloECLAMC: The Latin-American collaborative study of congenital malformations, Centro de Educación Médica e Investigaciones Clínicas (CEMIC-CONICET), Buenos Aires, Argentina.
ECEMC Peripheral Group
Karin KällénInstitute of Clinical Sciences, Lund University and National Board of Health and Welfare, Lund, Sweden.
Margery MorganCongenital anomaly register and information service, Public Health Wales, Cardiff, Wales, UK.
Wladimir WerteleckiOMNI-Net Ukraine Birth Defects Program, Rivne, Ukraine.
Amy NanceUtah Department of Health, Utah Birth Defect Network, Salt Lake City, Utah, USA.ORCID 0000-0002-4326-890X
Erin B StallingsDivision of Birth Defects and Infant Disorders, National Center on Birth Defects and Development Disabilities, US Centers for Disease Control and Prevention, Atlanta, Georgia, USA.
Wendy N NembhardDepartment of Epidemiology and The Arkansas Reproductive Health Monitoring System, University of Arkansas for Medical Sciences, Little Rock, Arkansas, USA.ORCID 0000-0002-8318-4049
Peter MosseySchool of Dentistry, University of Dundee, Dundee, Scotland, UK.
University of Dundee · GBCenters for Disease Control and Prevention · USNational Board of Health and Welfare · SEAdministración Nacional de Laboratorios e Institutos de Salud · ARConsejo Nacional de Investigaciones Científicas y Técnicas · ARFondazione IRCCS Istituto Nazionale dei Tumori · ITInserm · FRIstituto di Fisiologia Clinica · ITMinistry for Health · MTNational Institute of Genomic Medicine · MXOtto-von-Guericke-Universität Magdeburg · DEPontificia Universidad Javeriana · COPublic Health Wales · GBSlovak Medical University · SKSoroka Medical Center · ILTabriz University of Medical Sciences · IRThomayer University Hospital · CZUniversidad Autónoma de Nuevo León · MXUniversity Medical Center Groningen · NLUtah Department of Health · US

Funding

Intramural CDC HHS CC999999
6 · The paper itself

Abstract

backgroundCleft lip with cleft palate (CLP) is a congenital condition that affects both the oral cavity and the lips. This study estimated the prevalence and mortality of CLP using surveillance data collected from birth defect registries around the world.

methodsData from 22 population- and hospital-based surveillance programs affiliated with the International Clearinghouse for Birth Defects Surveillance and Research (ICBDSR) in 18 countries on live births (LB), stillbirths (SB), and elective terminations of pregnancy for fetal anomaly (ETOPFA) for CLP from 1974 to 2014 were analyzed. Prevalence and survival (survival for LB only) estimates were calculated for total and subclassifications of CLP and by pregnancy outcome.

resultsThe pooled prevalence of total CLP cases was 6.4 CLP per 10,000 births. The prevalence of CLP and all of the pregnancy outcomes varied across programs. Higher ETOPFA rates were recorded in most European programs compared to programs in other continents. In programs reporting low ETOPFA rates or where there was no ascertainment of ETOPFA, the rate of CLP among LB and SB was higher compared to those where ETOPFA rates were ascertained. Overall survival for total CLP was 91%. For isolated CLP, the survival was 97.7%. CLP associated with multiple congenital anomalies had an overall survival of 77.1%, and for CLP associated with genetic/chromosomal syndromes, overall survival was 40.9%.

conclusionsTotal CLP prevalence reported in this study is lower than estimates from prior studies, with variation by pregnancy outcomes between programs. Survival was lower when CLP was associated with other congenital anomalies or syndromes compared to isolated CLP.

Indexed as

Cleft LipCleft PalateFemaleHumansPregnancyPregnancy OutcomePrevalenceStillbirthSyndromecongenital anomaliescraniofacial abnormalitiesmortalityprevalencesurveillance

Identifiers

PMID37186369
PMCPMC10405782
OpenAlexW4367055833

What OpenQuestion holds

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LicenceCC BY
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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.