Evidence map›Paper›PMID 39085583›Full record

ReviewEuropean journal of human genetics : EJHG2024

Bardet-Biedl syndrome improved diagnosis criteria and management: Inter European Reference Networks consensus statement and recommendations.

Hélène Dollfus, Marc R Lilien, Pietro Maffei, Alain Verloes, Jean Muller, Giacomo M Bacci, Metin Cetiner, Erica L T van den Akker, Monika Grudzinska Pechhacker, Francesco Testa and 17 more

2 registry-linked trialsAbstract readReviewConsensus Statement
In one paragraph

Review in European journal of human genetics : EJHG, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to 2 registered trials, which are not on this map. Cited by 50 papers.

0numbers the graph read from it
0cells of the map it votes in
50citing 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.

NCT06615011 not yet recruitingnot on this map

Bardet Beidle Syndrome in a Syrian Adolescent : a Rare Case Report

Typeobservational_patient_registrySponsorAl Baath UniversityRan2024 to 2025Enrolled100ConditionsBardet Biedl Syndrome (BBS)
NCT07674290 phase4recruitingnot on this map

Real-World Effectiveness, Safety and Patient-reported Outcomes of Setmelanotide in Patients With Bardet-Biedl Syndrome: A Prospective Mono Centric Observational Interventional Study

TypeinterventionalSponsorTom HühneRan2023 to 2030Enrolled200ConditionsBardet Biedl Syndrome (BBS), Bardet Biedl Syndrome, Bardet-Biedl Syndrome (BBS), Alstrom SyndromeArmsSetmelanotide
3 · Its place in the literature

Who cites it

50 citing papers in PubMed.

  1. Trial
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  6. Review
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  13. Review
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  15. Review
  16. Bardet-Biedl syndrome in two sibling pairs: a case series.Journal of medical case reports · 2026
    Article
  17. Article
  18. Ciliary Defects in Inherited Retinal Diseases.Advanced genetics (Hoboken, N.J.) · 2026
    Review
  19. Article
  20. 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

27 authors.

Hélène Dollfus *ERN-EYE Centre de Référence Pour les Affections Rares en Génétique Ophtalmologique (CRMR CARGO), Institut de Génétique Médicale d'Alsace (IGMA), FSMR SENSGENE, Hôpitaux Universitaires de Strasbourg, Strasbourg, France. dollfus@unistra.fr.ORCID 0000-0002-2249-895X
Marc R Lilien *ERKNet Wilhelmina Children's Hospital, University Medical Center, Utrecht, The Netherlands.ORCID 0000-0002-8956-0324
Pietro Maffei *Endo-ERN Department of Medicine (DIMED), 3rd Medical Clinic, Padua University, Padua, Italy.
Alain Verloes *ERN-ITHACA Department of Genetics, AP-HP - Université de Paris; INSERM UMR 1141 "NeuroDiderot", Hôpital Robert Debré, Paris, France.ORCID 0000-0003-4819-0264
Jean Muller *Laboratoires de Diagnostic Génétique, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.ORCID 0000-0002-7682-559X
Giacomo M BacciERN-EYE Pediatric Ophthalmology Unit, Meyer Children's Hospital IRCCS, University of Florence, Florence, Italy.ORCID 0000-0002-8741-4833
Metin CetinerERKNet Children's Hospital, Pediatrics II, University of Essen, Essen, Germany.ORCID 0000-0002-0918-9204
Erica L T van den AkkerEndo-ERN Obesity Center CGG, Erasmus MC, University Medical Center Rotterdam, Division of Endocrinology, Department of Pediatrics, Erasmus MC-Sophia, University Medical Center Rotterdam, Rotterdam, The Netherlands.ORCID 0000-0001-5352-9328
Monika Grudzinska PechhackerERN-EYE Coordination Center, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
Francesco TestaERN-EYE Eye Clinic, Multidisciplinary Department of Medical, Surgical and Dental Sciences, University of Campania Luigi Vanvitelli, Naples, Italy.
Didier LacombeERN-ITHACA Department of Medical Genetics, CHU Bordeaux, INSERM Unit_1211, Laboratory "Rare Diseases: Genetics and Metabolism", University of Bordeaux, Bordeaux, France.
Marijn F StokmanERKNet Department of Human Genetics, Radboud University Medical Centre, Nijmegen, The Netherlands.
Francesca SimonelliERN-EYE Eye Clinic, Multidisciplinary Department of Medical, Surgical and Dental Sciences, University of Campania Luigi Vanvitelli, Naples, Italy.
Aurélie GouroncERN-EYE Centre de Référence Pour les Affections Rares en Génétique Ophtalmologique (CRMR CARGO), Institut de Génétique Médicale d'Alsace (IGMA), FSMR SENSGENE, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.ORCID 0000-0003-0758-8882
Amélie GavardERN-EYE Coordination Center, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
Mieke M van HaelstERN-ITHACA Department of Human Genetics, Section Clinical Genetics, Amsterdam UMC location University of Amsterdam, Amsterdam, The Netherlands.ORCID 0000-0002-7519-0246
Jens KoenigERKNet University Children's Hospital Muenster, Muenster, NRW, Germany.ORCID 0000-0002-9732-8656
Sylvie RossignolEndo-ERN Département de Pédiatrie, Hôpitaux Universitaires de Strasbourg, Strasbourg, France.
Carsten BergmannDepartment of Medicine IV, Faculty of Medicine, Medical Center, University of Freiburg, Freiburg, Germany.
Miriam ZacchiaERKNet Division of Nephrology, Department of Translational Medical Sciences, University of Campania "L. Vanvitelli", Naples, Italy.
Bart P LeroyERN-EYE Department of Ophthalmology & Department of Head & Skin, Ghent University Hospital and Ghent University, Ghent, Belgium.ORCID 0000-0002-9899-2081
Héléna MosbahEndo-ERN Department of Endocrinology, Diabetology & Nutrition, University Hospital of Poitiers, Poitiers, France.
Albertien M Van EerdeERKNet Department of Genetics, University Medical Center Utrecht, Utrecht, The Netherlands.ORCID 0000-0001-5953-5956
Djalila MekahliERKNet PKD Research Group, Department of Cellular and Molecular Medicine, KU Leuven, Leuven, Belgium.ORCID 0000-0003-0954-6088
Aude ServaisERKNet Department of Kidney and Metabolic Diseases, Transplantation and Clinical Immunology, Necker Hospital, AP-HP, Centre of Reference for the French Nationwide MARHEANetwork (CNR-MARHEA), Paris, France.
Christine PoitouEndo-ERN Centre de Référence pour les obésités rares (CRMR PRADORT), Assistance Publique Hôpitaux de Paris, Pitié-Salpêtrière Hospital, Sorbonne Université, INSERM, Nutrition & Obesities: Systemic Approaches Research Group (NutriOmics), Paris, France.
Diana ValverdeCINBIO, Universidad de Vigo, Grupo de Investigación en Enfermedades Raras, Instituto de Investigación Sanitaria Galicia Sur (IIS Galicia Sur), Vigo, Spain.ORCID 0000-0002-7024-1657

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Four European Reference Networks (ERN-EYE, ERKNet, Endo-ERN, ERN-ITHACA) have teamed up to establish a consensus statement and recommendations for Bardet-Biedl syndrome (BBS). BBS is an autosomal recessive ciliopathy with at least 26 genes identified to date. The clinical manifestations are pleiotropic, can be observed in utero and will progress with age. Genetic testing has progressively improved in the last years prompting for a revision of the diagnostic criteria taking into account clinical Primary and Secondary features, as well as positive or negative molecular diagnosis. This consensus statement also emphasizes on initial diagnosis, monitoring and lifelong follow-up, and symptomatic care that can be provided to patients and family members according to the involved care professionals. For paediatricians, developmental anomalies can be at the forefront for diagnosis (such as polydactyly) but can require specific care, such as for associated neuro developmental disorders. For ophthalmology, the early onset retinal degeneration requires ad hoc functional and imaging technologies and specific care for severe visual impairment. For endocrinology, among other manifestations, early onset obesity and its complications has benefited from better evaluation of eating behaviour problems, improved lifestyle programs, and from novel pharmacological therapies. Kidney and urinary track involvements warrants lifespan attention, as chronic kidney failure can occur and early management might improve outcome. This consensus recommends revised diagnostic criteria for BBS that will ensure certainty of diagnosis, giving robust grounds for genetic counselling as well as in the perspective of future trials for innovative therapies.

Indexed as

Bardet-Biedl SyndromeEuropeGenetic TestingHumans

Identifiers

PMID39085583
PMCPMC11576898

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Registered trials

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.