| Literature DB >> 33654309 |
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.Entities:
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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