Evidence map›Paper›PMID 38671254›Full record

ArticleEuropean journal of epidemiology2024

A multidisciplinary and structured investigation of three suspected clusters of transverse upper limb reduction defects in France.

Julie Boudet-Berquier, Christophe Demattei, Laurence Guldner, Anne Gallay, Sylvie Manouvrier, Jérémie Botton, Claire Philippat, Fleur Delva, Juliette Bloch, Caroline Semaille and 29 more

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

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1 · What the graph read from it

What it found

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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

39 authors.

Julie Boudet-BerquierNon-Communicable Diseases and Injuries Department, Santé Publique France, Saint Maurice, France. Julie.BOUDET-BERQUIER2@santepubliquefrance.fr.ORCID http://orcid.org/0000-0002-2278-3697
Christophe DematteiDepartment of Biostatistics, Clinical Epidemiology, Public Health and Innovation in Methodology (BESPIM), Centre Hospitalier Universitaire de Nîmes, Nîmes, Languedoc-Roussillon, France.
Laurence GuldnerEnvironment and Work Department, Santé Publique France, Saint Maurice, Health, France.
Anne GallayNon-Communicable Diseases and Injuries Department, Santé Publique France, Saint Maurice, France.
Sylvie ManouvrierReference Centre of Developmental Anomalies and Defect Syndromes, RADEME Maladies Rares du Développement Et du Métabolisme, Université Lille, 7364, Lille, EA, France.
Jérémie BottonEPI-PHARE Scientific Interest Group in Epidemiology of Health Products from the French National Agency for the Safety of Medicines and Health Products and the French National Health Insurance, Saint-Denis, France.
Claire PhilippatInstitute for Advanced Biosciences, INSERM U1209, CNRS UMR 5309, University Grenoble Alpes, Grenoble, France.
Fleur DelvaINSERM, BPH, UMR1219, EPICENE Team, University of Bordeaux, Bordeaux, France.
Juliette BlochANSES, Maisons-Alfort, France.
Caroline SemailleExecutive Department, Santé Publique France, Saint-Maurice, France.
Sylvie OdentReference Centre of Developmental Anomalies and Defect Syndromes, Hôpital Sud, CHU de Rennes, Rennes, France.
Isabelle PerthusAuvergne registry of congenital anomalies (CEMC-Auvergne), Department of clinical genetics, Centre de Référence des Maladies Rares, University Hospital of Clermont-Ferrand, Clermont-Ferrand, France.
Hanitra RandrianaivoLa Réunion registry of congenital anomalies, Centre Hospitalier Universitaire de La Réunion, Île de la Réunion, France.
Sylvie BabajkoMolecular Oral Pathophysiology Laboratory, Centre de Recherche des Cordeliers, INSERM UMRS, Université Paris Cité, Sorbonne Université, 1138, Paris, France.
Tiphaine BarjatDepartment of Gynecology and Obstetrics, Centre Hospitalier Universitaire de Saint-Étienne, Saint-Étienne, France.
Claire BeneteauDepartment of Human Genetics, University Hospital Hôtel Dieu, Nantes, France.
Naima BrennetotFrench National Reference for Children Limb Defect, Hôpitaux de Saint-Maurice, Saint Maurice, France.
Ester GarneDepartment of Pediatrics and Adolescent Medicine, Lillebaelt Hospital, University Hospital of Southern Denmark, Kolding, Denmark.
Georges HaddadCH Simone Veil, Blois, France.
Mounia HocineConservatoire National des Arts et Métiers, Paris, France.
Isabelle LacroixREGARDs Network, Pharmacologie Médicale et Clinique, CERPOP INSERM UMR 1295-SPHERE team, Faculté de Médecine Université de Toulouse, Centre Hospitalier Universitaire de Toulouse, Toulouse, France.
Klervi LeuraudInstitute for Radiological Protection and Nuclear Safety (IRSN), Fontenay-aux-Roses, France.
Michel MenchINRAE, BIOGECO, UMR 1202, University of Bordeaux, F‑33615, Pessac, France.
Joan MorrisPopulation Health Research Institute, St George's University of London, London, UK.
Sophie PatrierDepartment of Pathology, Rouen University Hospital, Rouen, France.
Arnaud SarteletClinical Department of Ruminant, University of Liège, 4000, Liège, Belgium.
Alain VerloesGenetics Department, APHP, Robert-Debré University Hospital, Paris, France.
Christophe BonaldiMethodology and Biostatistics Department, Santé Publique France, Saint Maurice, France.
Mélina Le BarbierEnvironment and Work Department, Santé Publique France, Saint Maurice, Health, France.
Bertrand GagnièreCellule Bretagne, Santé Publique France, Rennes, France.
Philippe PépinCellule Auvergne-Rhône-Alpes, Santé Publique France, Clermont-Ferrand, France.
Ronan OllivierCellule Pays-de-la Loire, Santé Publique France,, Nantes, France.
Monique BitounCellule Pays-de-la Loire, Santé Publique France,, Nantes, France.
Lisa KingCellule Pays-de-la Loire, Santé Publique France,, Nantes, France.
Andrea Guajardo-VillarMethodology and Biostatistics Department, Santé Publique France, Saint Maurice, France.
Eugenia GomesNon-Communicable Diseases and Injuries Department, Santé Publique France, Saint Maurice, France.
Jean-Claude DesenclosANSES, Maisons-Alfort, France.
Nolwenn RegnaultNon-Communicable Diseases and Injuries Department, Santé Publique France, Saint Maurice, France.
Alexandra BenachiDepartment of Obstetrics and Gynecology, DMU Santé des femmes et des nouveau-nés, Hopital Antoine Béclère, Assistance Publique-Hôpitaux de Paris, Université Paris Saclay, Clamart, France.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

introductionBetween 2019-2021, facing public concern, a scientific expert committee (SEC) reanalysed suspected clusters of transverse upper limb reduction defects (TULRD) in three administrative areas in France, where initial investigations had not identified any risk exposure. We share here the national approach we developed for managing suspicious clusters of the same group of congenital anomalies occurring in several areas.

methodsThe SEC analysed the medical records of TURLD suspected cases and performed spatiotemporal analyses on confirmed cases. If the cluster was statistically significant and included at least three cases, the SEC reviewed exposures obtained from questionnaires, environmental databases, and a survey among farmers living near to cases' homes concerning their plant product use.

resultsAfter case re-ascertainment, no statistically significant cluster was observed in the first administrative areas. In the second area, a cluster of four children born in two nearby towns over two years was confirmed, but as with the initial investigations, no exposure to a known risk factor explaining the number of cases in excess was identified. In the third area, a cluster including just two cases born the same year in the same town was confirmed. DISCUSSION: Our experience highlights that in the event of suspicious clusters occurring in different areas of a country, a coordinated and standardised approach should be preferred.

Indexed as

Upper Extremity Deformities, CongenitalChildCluster AnalysisEnvironmental ExposureFemaleFranceHumansInfantMaleRisk FactorsSpatio-Temporal AnalysisUpper ExtremityBirth defectsClusterCongenital anomaliesEnvironmentLimb reduction defectsSpatiotemporal analyses

Identifiers

PMID38671254

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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.