Literature DB >> 33654309

MCM complex members MCM3 and MCM7 are associated with a phenotypic spectrum from Meier-Gorlin syndrome to lipodystrophy and adrenal insufficiency.

Karen M Knapp1, Danielle E Jenkins1, Rosie Sullivan1, Frederike L Harms2, Leonie von Elsner2, Charlotte W Ockeloen3, Sonja de Munnik3, Ernie M H F Bongers3, Jennie Murray4,5, Nicholas Pachter6,7, Jonas Denecke8, Kerstin Kutsche2, Louise S Bicknell9.   

Abstract

The MCM2-7 helicase is a heterohexameric complex with essential roles as part of both the pre-replication and pre-initiation complexes in the early stages of DNA replication. Meier-Gorlin syndrome, a rare primordial dwarfism, is strongly associated with disruption to the pre-replication complex, including a single case described with variants in MCM5. Conversely, a biallelic pathogenic variant in MCM4 underlies immune deficiency with growth retardation, features also seen in individuals with pathogenic variants in other pre-initiation complex encoding genes such as GINS1, MCM10, and POLE. Through exome and chromium genome sequencing, supported by functional studies, we identify biallelic pathogenic variants in MCM7 and a strong candidate biallelic pathogenic variant in MCM3. We confirm variants in MCM7 are deleterious and through interfering with MCM complex formation, impact efficiency of S phase progression. The associated phenotypes are striking; one patient has typical Meier-Gorlin syndrome, whereas the second case has a multi-system disorder with neonatal progeroid appearance, lipodystrophy and adrenal insufficiency. We provide further insight into the developmental complexity of disrupted MCM function, highlighted by two patients with a similar variant profile in MCM7 but disparate clinical features. Our results build on other genetic findings linked to disruption of the pre-replication and pre-initiation complexes, and the replisome, and expand the complex clinical genetics landscape emerging due to disruption of DNA replication.
© 2021. The Author(s), under exclusive licence to European Society of Human Genetics.

Entities:  

Mesh:

Substances:

Year:  2021        PMID: 33654309      PMCID: PMC8298597          DOI: 10.1038/s41431-021-00839-4

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   5.351


  44 in total

1.  Isolation and characterization of various complexes of the minichromosome maintenance proteins of Schizosaccharomyces pombe.

Authors:  J K Lee; J Hurwitz
Journal:  J Biol Chem       Date:  2000-06-23       Impact factor: 5.157

2.  Meier-Gorlin syndrome: growth and secondary sexual development of a microcephalic primordial dwarfism disorder.

Authors:  Sonja A de Munnik; Barto J Otten; Jeroen Schoots; Louise S Bicknell; Salim Aftimos; Jumana Y Al-Aama; Yolande van Bever; Michael B Bober; George F Borm; Jill Clayton-Smith; Cheri L Deal; Alaa Y Edrees; Murray Feingold; Alan Fryer; Johanna M van Hagen; Raoul C Hennekam; Maaike C E Jansweijer; Diana Johnson; Sarina G Kant; John M Opitz; A Radha Ramadevi; Willie Reardon; Alison Ross; Pierre Sarda; Constance T R M Schrander-Stumpel; A Erik Sluiter; I Karen Temple; Paulien A Terhal; Annick Toutain; Carol A Wise; Michael Wright; David L Skidmore; Mark E Samuels; Lies H Hoefsloot; Nine V A M Knoers; Han G Brunner; Andrew P Jackson; Ernie M H F Bongers
Journal:  Am J Med Genet A       Date:  2012-09-28       Impact factor: 2.802

3.  The dynamics of eukaryotic replication initiation: origin specificity, licensing, and firing at the single-molecule level.

Authors:  Daniel Duzdevich; Megan D Warner; Simina Ticau; Nikola A Ivica; Stephen P Bell; Eric C Greene
Journal:  Mol Cell       Date:  2015-04-23       Impact factor: 17.970

4.  Selective bypass of a lagging strand roadblock by the eukaryotic replicative DNA helicase.

Authors:  Yu V Fu; Hasan Yardimci; David T Long; The Vinh Ho; Angelo Guainazzi; Vladimir P Bermudez; Jerard Hurwitz; Antoine van Oijen; Orlando D Schärer; Johannes C Walter
Journal:  Cell       Date:  2011-09-16       Impact factor: 41.582

5.  Depletion of minichromosome maintenance protein 5 in the zebrafish retina causes cell-cycle defect and apoptosis.

Authors:  Soojin Ryu; Jochen Holzschuh; Simone Erhardt; Anne-Kathrin Ettl; Wolfgang Driever
Journal:  Proc Natl Acad Sci U S A       Date:  2005-12-08       Impact factor: 11.205

6.  Geminin is required for Hox gene regulation to pattern the developing limb.

Authors:  Emily M A Lewis; Savita Sankar; Caili Tong; Ethan S Patterson; Laura E Waller; Paul Gontarz; Bo Zhang; David M Ornitz; Kristen L Kroll
Journal:  Dev Biol       Date:  2020-05-23       Impact factor: 3.582

7.  Polymerase ε1 mutation in a human syndrome with facial dysmorphism, immunodeficiency, livedo, and short stature ("FILS syndrome").

Authors:  Jana Pachlopnik Schmid; Roxane Lemoine; Nadine Nehme; Valéry Cormier-Daire; Patrick Revy; Franck Debeurme; Marianne Debré; Patrick Nitschke; Christine Bole-Feysot; Laurence Legeai-Mallet; Annick Lim; Jean-Pierre de Villartay; Capucine Picard; Anne Durandy; Alain Fischer; Geneviève de Saint Basile
Journal:  J Exp Med       Date:  2012-12-10       Impact factor: 14.307

8.  Mutations in DONSON disrupt replication fork stability and cause microcephalic dwarfism.

Authors:  John J Reynolds; Louise S Bicknell; Paula Carroll; Martin R Higgs; Ranad Shaheen; Jennie E Murray; Dimitrios K Papadopoulos; Andrea Leitch; Olga Murina; Žygimantė Tarnauskaitė; Sarah R Wessel; Anastasia Zlatanou; Audrey Vernet; Alex von Kriegsheim; Rachel M A Mottram; Clare V Logan; Hannah Bye; Yun Li; Alexander Brean; Sateesh Maddirevula; Rachel C Challis; Kassiani Skouloudaki; Agaadir Almoisheer; Hessa S Alsaif; Ariella Amar; Natalie J Prescott; Michael B Bober; Angela Duker; Eissa Faqeih; Mohammed Zain Seidahmed; Saeed Al Tala; Abdulrahman Alswaid; Saleem Ahmed; Jumana Yousuf Al-Aama; Janine Altmüller; Mohammed Al Balwi; Angela F Brady; Luciana Chessa; Helen Cox; Rita Fischetto; Raoul Heller; Bertram D Henderson; Emma Hobson; Peter Nürnberg; E Ferda Percin; Angela Peron; Luigina Spaccini; Alan J Quigley; Seema Thakur; Carol A Wise; Grace Yoon; Maha Alnemer; Pavel Tomancak; Gökhan Yigit; A Malcolm R Taylor; Martin A M Reijns; Michael A Simpson; David Cortez; Fowzan S Alkuraya; Christopher G Mathew; Andrew P Jackson; Grant S Stewart
Journal:  Nat Genet       Date:  2017-02-13       Impact factor: 38.330

9.  Mutations in CDC45, Encoding an Essential Component of the Pre-initiation Complex, Cause Meier-Gorlin Syndrome and Craniosynostosis.

Authors:  Aimee L Fenwick; Maciej Kliszczak; Fay Cooper; Jennie Murray; Luis Sanchez-Pulido; Stephen R F Twigg; Anne Goriely; Simon J McGowan; Kerry A Miller; Indira B Taylor; Clare Logan; Sevcan Bozdogan; Sumita Danda; Joanne Dixon; Solaf M Elsayed; Ezzat Elsobky; Alice Gardham; Mariette J V Hoffer; Marije Koopmans; Donna M McDonald-McGinn; Gijs W E Santen; Ravi Savarirayan; Deepthi de Silva; Olivier Vanakker; Steven A Wall; Louise C Wilson; Ozge Ozalp Yuregir; Elaine H Zackai; Chris P Ponting; Andrew P Jackson; Andrew O M Wilkie; Wojciech Niedzwiedz; Louise S Bicknell
Journal:  Am J Hum Genet       Date:  2016-06-30       Impact factor: 11.025

10.  Zebrafish cdc6 hypomorphic mutation causes Meier-Gorlin syndrome-like phenotype.

Authors:  Likun Yao; Jing Chen; Xiaotong Wu; Shunji Jia; Anming Meng
Journal:  Hum Mol Genet       Date:  2017-11-01       Impact factor: 6.150

View more
  7 in total

Review 1.  Efficiency and equity in origin licensing to ensure complete DNA replication.

Authors:  Liu Mei; Jeanette Gowen Cook
Journal:  Biochem Soc Trans       Date:  2021-11-01       Impact factor: 4.919

2.  Hypomorphic GINS3 variants alter DNA replication and cause Meier-Gorlin syndrome.

Authors:  Mary E McQuaid; Kashif Ahmed; Stephanie Tran; Justine Rousseau; Ranad Shaheen; Kristin D Kernohan; Kyoko E Yuki; Prerna Grover; Ema S Dreseris; Sameen Ahmed; Lucie Dupuis; Jennifer Stimec; Mary Shago; Zuhair N Al-Hassnan; Roch Tremblay; Philipp G Maass; Michael D Wilson; Eyal Grunebaum; Kym M Boycott; François-Michel Boisvert; Sateesh Maddirevula; Eissa A Faqeih; Fahad Almanjomi; Zaheer Ullah Khan; Fowzan S Alkuraya; Philippe M Campeau; Peter Kannu; Eric I Campos; Hugo Wurtele
Journal:  JCI Insight       Date:  2022-05-23

3.  Clinical genetics-it's polygenic.

Authors:  Alisdair McNeill
Journal:  Eur J Hum Genet       Date:  2021-07       Impact factor: 5.351

4.  Validation of a high throughput screening assay to identify small molecules that target the eukaryotic replicative helicase.

Authors:  Jordan Sanders; Michael Castiglione; Tongying Shun; Laura L Vollmer; Mark E Schurdak; Andreas Vogt; Anthony Schwacha
Journal:  SLAS Discov       Date:  2022-01-08       Impact factor: 3.341

5.  Novel Compound Heterozygous Variants in the CDC6 Gene in a Russian Patient with Meier-Gorlin Syndrome.

Authors:  Viktoriia Zabnenkova; Olga Shchagina; Olga Makienko; Galina Matyushchenko; Oxana Ryzhkova
Journal:  Appl Clin Genet       Date:  2022-01-06

6.  Meier-Gorlin Syndrome: Clinical Misdiagnosis, Genetic Testing and Functional Analysis of ORC6 Mutations and the Development of a Prenatal Test.

Authors:  Maria S Nazarenko; Iuliia V Viakhireva; Mikhail Y Skoblov; Elena V Soloveva; Aleksei A Sleptcov; Ludmila P Nazarenko
Journal:  Int J Mol Sci       Date:  2022-08-17       Impact factor: 6.208

Review 7.  Molecular and Cellular Bases of Lipodystrophy Syndromes.

Authors:  Jamila Zammouri; Camille Vatier; Emilie Capel; Martine Auclair; Caroline Storey-London; Elise Bismuth; Héléna Mosbah; Bruno Donadille; Sonja Janmaat; Bruno Fève; Isabelle Jéru; Corinne Vigouroux
Journal:  Front Endocrinol (Lausanne)       Date:  2022-01-03       Impact factor: 5.555

  7 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.