Evidence map›Paper›PMID 39837771›Full record

ReviewClinical genetics2025

The Phenotypic and Genotypic Spectrum of BRPF1-Related Disorder: 29 New Patients and Literature Review.

Cindy Colson, Marine Tessarech, Elise Boucher-Brischoux, Odile Boute-Benejean, Catherine Vincent-Delorme, Clémence Vanlerberghe, Simon Boussion, Justine Le Cunff, Bénédicte Duban-Bedu, Laurence Faivre and 12 more

Abstract readReview
In one paragraph

Review in Clinical genetics, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing 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

3 citing papers in PubMed.

  1. Article
  2. Article
  3. 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

22 authors.

Cindy ColsonUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Marine TessarechUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Elise Boucher-BrischouxCHU Besançon, Centre de Génétique Humaine, Besançon, France.ORCID 0000-0002-4816-1431
Odile Boute-BenejeanUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Catherine Vincent-DelormeUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Clémence VanlerbergheUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Simon BoussionUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.ORCID 0009-0000-0919-7207
Justine Le CunffUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Bénédicte Duban-BeduHôpital Saint Vincent de Paul, GHICL, Centre de Génétique Chromosomique, Lille, France.
Laurence FaivreUMR1231 GAD, Inserm, Université Bourgogne-Franche Comté, Dijon, France.
Christel ThauvinUMR1231 GAD, Inserm, Université Bourgogne-Franche Comté, Dijon, France.
Christophe PhilippeCHU Dijon Bourgogne, Service de Génomique Médicale, Laboratoire de Biologie Médicale, Dijon, France.
Ange-Line BruelCHU Dijon Bourgogne, Service de Génomique Médicale, Laboratoire de Biologie Médicale, Dijon, France.ORCID 0000-0002-0526-465X
Frédéric Tran Mau-ThemCHU Dijon Bourgogne, Service de Génomique Médicale, Laboratoire de Biologie Médicale, Dijon, France.
Clara HoudayerCHU Angers, Service de Génétique Médicale, Angers, France.
Gaetan LescaHospices Civils de Lyon, Service de Génétique, Université Claude Bernard Lyon 1, Lyon, France.
Audrey PutouxHospices Civils de Lyon, Service de Génétique, Université Claude Bernard Lyon 1, Lyon, France.
Jonathan LévyDépartement de Génétique, Hôpital Universitaire Robert-Debré, Paris, France.ORCID 0000-0002-8822-816X
Olivier PatatDépartement de Génétique médicale, CHU Toulouse, Toulouse, France.
Marlène RioService de génétique, Hôpital Necker, Paris, France.
Jamal GhoumidUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.
Thomas SmolUniv. Lille, CHU Lille, ULR7364 - RADEME - Maladies RAres du DEveloppement embryonnaire et du Métabolisme, CRMR Déficiences Intellectuelles de Causes Rares, Lille, France.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Intellectual Developmental Disorder with Dysmorphic Facies and Ptosis (IDDDFP) is a rare autosomal dominant syndrome caused by pathogenic variants in the BRPF1 gene, which is critical for chromatin regulation. This study expands the clinical and molecular spectrum of IDDDFP by analysing 29 new patients from 20 families with confirmed BRPF1 variants. Our cohort presented with a wide range of clinical features including developmental delay, intellectual disability (ID) and characteristic dysmorphic facial features such as ptosis, blepharophimosis and a broad nasal bridge. New phenotypic features identified include palpebral oedema, laterally elongated eyebrows, low hanging columella and hypertrichosis. Neuropsychological assessment reveals a predominance of mild to moderate ID, with cognitive profiles showing variability in verbal and visual processing. Structural abnormalities such as agenesis of the corpus callosum and ocular defects were noted, consistent with previous studies but with some differences. Familial analysis revealed variability in clinical expression. Our findings highlight the diverse clinical manifestations of BRPF1-related disorders and suggest that comprehensive ophthalmological evaluation is essential for the management of these patients.

Indexed as

Adaptor Proteins, Signal TransducingBlepharoptosisDevelopmental DisabilitiesIntellectual DisabilityAdolescentAdultChildChild, PreschoolFaciesFemaleGenetic Association StudiesGenotypeHumansInfantMaleMutationAdaptor Proteins, Signal TransducingBRPF1IDDDFPneurodevelopmental disordersphenotypic variability

Identifiers

PMID39837771
PMCPMC11973018

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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.