ArticleNucleus (Austin, Tex.)
Dosage effect of zero to three functional LBR-genes in vivo and in vitro.
Article in Nucleus (Austin, Tex.). The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 16 papers.
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Who cites it
16 citing papers in PubMed, 20 citations in OpenAlex.
- A homozygous lamin B receptor variant resulting in Pelger-Huët anomaly without skeletal dysplasia.Nucleus (Austin, Tex.) · 2026Article
- Shaping neutrophil morphology and function: the importance of a segmented nucleus.Nature reviews. Immunology · 2026Review
- Envelope-Limited Chromatin Sheets (ELCS) Formation in The Nuclear Envelope of HL-60/S4 Cells.bioRxiv : the preprint server for biology · 2026Article
- Polarization increases nuclear stiffness in macrophages despite reduction in lamin A/C levels.npj biological physics and mechanics · 2026Article
- Nuclear tension controls mitotic entry by regulating cyclin B1 nuclear translocation.The Journal of cell biology · 2022Article
- Nuclear Morphological Abnormalities in Cancer: A Search for Unifying Mechanisms.Results and problems in cell differentiation · 2022Article
- Microtubules and Dynein Regulate Human Neutrophil Nuclear Volume and Hypersegmentation DuringFrontiers in immunology · 2021Article
- Article
- The nucleus acts as a ruler tailoring cell responses to spatial constraints.Science (New York, N.Y.) · 2020Article
- 1q42.12q42.2 Deletion in a Child with Midline Defects and Hypoplasia of the Corpus Callosum.Molecular syndromology · 2019Article
- The Neutrophil Nucleus: An Important Influence on Neutrophil Migration and Function.Frontiers in immunology · 2018Review
- Psychomotor retardation with a 1q42.11-q42.12 deletion.Hereditas · 2017Article
- Molecular evolution of psbA gene in ferns: unraveling selective pressure and co-evolutionary pattern.BMC evolutionary biology · 2012Article
- Nuclear mechanics in disease.Annual review of biomedical engineering · 2011Review
- An in vitro model for Pelger-Huët anomaly: stable knockdown of lamin B receptor in HL-60 cells.Nucleus (Austin, Tex.)Article
- Intranuclear membranes induced by lipidated proteins are derived from the nuclear envelope.Nucleus (Austin, Tex.)Article
Corrections and comments
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Authors and funding
13 authors at 5 institutions in 7 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The Lamin B receptor (LBR) is a pivotal architectural protein in the nuclear envelope. Mutations in the Lamin B receptor lead to nuclear hyposegmentation (Pelger-Huët anomaly). We have exactly quantified the nuclear lobulation in neutrophils from individuals with 0, 1, 2 and 3 functional copies of the lamin B receptor gene and analyzed the effect of different mutation types. Our data demonstrate that there is a highly significant gene-dosage effect between the gene copy number and the nuclear segmentation index of neutrophils. This finding is paralleled by a dose-dependent increase in LBR protein and staining intensity of the nuclear membrane in corresponding lymphoblastoid cell lines, which demonstrates a significant correlation on the protein level as well. We further show that LBR expression continually increases during granulopoiesis in vitro from human precursor cells with ovoid nuclei to multi-segmented neutrophil nuclei 11 days later, indicating relevance for regular human granulopoiesis. Altogether, LBR is a unique model that will allow the systematic study of gene-dosage effects and of modifying endogeneous and exogeneous factors on granulopoiesis.
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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.