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.

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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
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article
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article

WWOX-Related Epileptic Encephalopathy (WOREE Syndrome): Clinical Case Study and Literature Review

Annamaria Sapuppo, Vincenzo Sortino, Roberta Rocca, Xena Giada Pappalardo et al.
Current Issues in Molecular Biology
Genetics and Neurodevelopmental Disorders
article

WWOX-Related Epileptic Encephalopathy (WOREE Syndrome): Clinical Case Study and Literature Review

Annamaria Sapuppo, Vincenzo Sortino, Roberta Rocca, Xena Giada Pappalardo, Martino Ruggieri, Raffaele Falsaperla, Roberta Rizzo, Gaia Fusto
article en

Abstract

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.

Current Issues in Molecular BiologyVol. 48(5)
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)
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Openalex Percentile: Top 5%
Genetics and Neurodevelopmental Disorders
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