Department of Pathology, Dunedin School of Medicine, University of Otago, Dunedin, New Zealand.
Institute of Human Genetics, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.
Eur J Hum Genet. 2021 Jul;29(7):1110-1120. doi: 10.1038/s41431-021-00839-4. Epub 2021 Mar 2.
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.
MCM2-7 解旋酶是一个异六聚体复合物,在 DNA 复制早期作为前复制和起始前复合物的一部分具有重要作用。Meier-Gorlin 综合征是一种罕见的原始侏儒症,与前复制复合物的破坏密切相关,包括一例描述为 MCM5 变异的病例。相反,MCM4 的双等位基因致病性变异导致免疫缺陷伴生长迟缓,在其他起始前复合物编码基因(如 GINS1、MCM10 和 POLE)的致病性变异个体中也可见到这些特征。通过外显子组和铬基因组测序,并通过功能研究支持,我们确定了 MCM7 的双等位基因致病性变异和 MCM3 的一个强烈候选双等位基因致病性变异。我们证实 MCM7 中的变异是有害的,并通过干扰 MCM 复合物的形成,影响 S 期进展的效率。相关表型引人注目;一名患者具有典型的 Meier-Gorlin 综合征,而另一名患者则表现为新生儿早衰样、脂肪营养不良和肾上腺功能不全的多系统疾病。我们通过两名患者在 MCM7 中具有相似的变异谱但具有不同的临床特征,进一步深入了解了 MCM 功能中断的发育复杂性。我们的结果建立在其他与前复制和起始前复合物以及复制体破坏相关的遗传发现的基础上,并扩展了由于 DNA 复制中断而出现的复杂临床遗传学景观。