WWOX-Related Epileptic Encephalopathy (WOREE Syndrome): Clinical Case Study and Literature Review
The WW domain-containing oxidoreductase (WWOX) gene, well-known as a tumor suppressor, also has a crucial role as a transcription factor in the developing brain. The bi-allelic loss of the WWOX gene causes a condition characterized by drug-resistant epilepsy, developmental delay, and neurological impairments, often resulting in mortality within the first year of life, known as WWOX-related epileptic encephalopathy (WOREE) syndrome (MIM: 616211). Whole Exome Sequencing (WES) analysis was performed on a female patient who died within three months of birth and was diagnosed with microcephaly, severe early-onset refractory seizures, and drug-resistant epileptic encephalopathy. WES revealed a 38 kb CNV deletion spanning WWOX exons 6–7, and a known frameshift variant in exon 8, impairing a highly clinically significant region of the encoded protein. Clinical and genetic features of reported WOREE patients with WWOX gene deletions similar to our patient were analyzed. Our case highlights the clinical heterogeneity of WWOX variants in WOREE syndrome and expands the spectrum of reported compound heterozygous deletions. Further research needs to elucidate WWOX pathophysiology and improve diagnostic and therapeutic strategies.
Authors
- Annamaria Sapuppo (ORCID: https://orcid.org/0000-0001-9433-026X)
- Vincenzo Sortino (ORCID: https://orcid.org/0000-0002-0188-8430)
- Roberta Rocca (ORCID: https://orcid.org/0000-0001-9017-8088)
- Xena Giada Pappalardo (ORCID: https://orcid.org/0000-0002-4640-6677)
- Martino Ruggieri (ORCID: https://orcid.org/0000-0002-2658-4249)
- Raffaele Falsaperla (ORCID: https://orcid.org/0000-0002-4482-3506)
- Roberta Rizzo (ORCID: https://orcid.org/0000-0001-9507-9126)
- Gaia Fusto
Institutions
- University of Ferrara (IT)
- Università degli Studi di Enna Kore (IT)
- University of Catania (IT)
- Policlinico Universitario di Catania (IT)
- Institute for Biomedical Research and Innovation (IT)
- Azienda Ospedaliero Universitaria Policlinico "G.Rodolico - San Marco" (IT)
Publication Details
- Journal
- Current Issues in Molecular Biology
- Published
- 2026-04-25
- DOI
- https://doi.org/10.3390/cimb48050449
- Primary Topic
- Genetics and Neurodevelopmental Disorders
- Type
- article
- Field-Weighted Citation Impact
- 0.00