A Novel Heterozygous De Novo MORC2 Missense Variant Causes an Early Onset and Severe Neurodevelopmental Disorder
Daniel Arbide1, Nour Elkhateeb2, Ewa Goljan3
1Edinburgh Medical School, University of Edinburgh, Edinburgh, UK.
Abstract:
Microrchidia CW-type zinc finger protein 2 (MORC2) is an ATPase-containing nuclear protein which regulates transcription through chromatin remodelling and epigenetic silencing. MORC2 may have a role in the development of neurones, and dominant variants in this gene have recently been linked with disorders including Charcot-Marie-Tooth type 2Z disease, spinal muscular atrophy and, more recently, a neurodevelopmental syndrome consisting of developmental delay, impaired growth, dysmorphic facies, and axonal neuropathy (DIGFAN), presenting with hypotonia, microcephaly, brain atrophy, intellectual disability, hearing loss, faltering growth, and craniofacial dysmorphism. Notably, variants in MORC2 have shown clinical features overlapping with those of Cockayne and Leigh syndromes. Here, we report a case of MORC2-related DIGFAN syndrome in a female infant caused by a novel heterozygous de novo variant. The condition was early onset and severe, further expanding the range of genotypes associated with this disorder. Clinical features included unilateral hearing loss, developmental delay and regression within the first year of life, microcephaly, severe feeding difficulties, and faltering growth, resulting in death at 13 months of age.
Insights
Microrchidia CW-type zinc finger protein 2 (MORC2) gene variants cause DIGFAN syndrome, a severe neurodevelopmental disorder. This report details a novel MORC2 variant in an infant, expanding the known genetic spectrum and highlighting early-onset, severe clinical features.
Area of Science:
- Genetics
- Neuroscience
- Molecular Biology
Background:
- Microrchidia CW-type zinc finger protein 2 (MORC2) is a nuclear protein involved in transcription regulation via chromatin remodeling and epigenetic silencing.
- MORC2 plays a role in neuronal development, and its dominant variants are associated with various neurological disorders, including Charcot-Marie-Tooth disease, spinal muscular atrophy, and DIGFAN syndrome.
- DIGFAN syndrome is a neurodevelopmental disorder characterized by developmental delay, impaired growth, dysmorphic features, and axonal neuropathy, with overlapping features with Cockayne and Leigh syndromes.
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